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A 71-year-old African American female with no significant past medical history presented to her primary care physician in September 2020 for progressive swelling in her right parotid region for 3 months associated with a recent onset of dental pain. She denied any previous history of tobacco use or prior radiation exposure. Antibiotics resolved her dental pain without improvement of the right parotid swelling. She was then evaluated by head and neck surgery. Computed tomography (CT) of the head and neck revealed a 1.9 cm heterogeneously enhancing lesion in the anterior, superior superficial parotid gland without any enlarged nodes. She did not have any preoperative facial nerve involvement or lymphadenopathy. She underwent a total parotidectomy with clinically uninvolved margins. The complete facial nerve was dissected free of the neoplasm with gross preservation of function. Postoperatively, she had some weakness in the marginal mandibular branch of the facial nerve, along with weakness in closing her superior palpebral fissure of the right eye. Pathology of resected lesions revealed salivary duct carcinoma that was 1.7 cm in maximum dimension, positive for cytokeratin 7, HER2/neu equivocal 2+, and androgen receptor-positive. The tumor was negative for p40, mucin, estrogen receptor, progesterone receptor, TTF-1, SOX100, mammaglobin, p63, and s-100. HER2/neu was 1+ FISH not amplified. She had no lymphovascular invasion, but perineural invasion was present. Postoperative positron emission tomography (PET) scan before radiation was negative for metastatic disease. The tumor was staged pathologically as T1N0M0 stage I. She received adjuvant radiotherapy incorporating margin positivity with 6600 cGy in 30 fractions to the entire parotid bed and the adjacent nodes with 6000 cGy in 30 fractions. The remainder of her right neck received 5400 cGy in 30 fractions. She completed radiotherapy in January 2021. She developed facial dermatitis and dysgeusia as a result of radiation, but otherwise tolerated therapy. Currently, she is being followed with CT scans every 3 months and is without evidence of disease recurrence to date. |
A 45-year old woman presented with a huge left breast mass that had necrosis and a foul odor. She had a history of contralateral metaplastic carcinoma, and she had undergone a right modified radical mastectomy 15 years previously. She also presented with a 2-cm-sized painless palpable breast mass in her left breast two years before presentation for the case in this report, but she did not undergo any clinical work-up at that time. Then, approximately 2 or 3 months prior to presentation of this case, she noticed a mass extrusion of the outside skin with rapid growth. On physical examination, a huge necrotic mass 20 cm in diameter was seen in the entire left breast. The mass was extruded skin outside, and skin overlying the mass was thickened with edematous change (Figure ). The axilla showed multiple conglomerated lymph nodes (LNs) that were fixed to the chest wall.\nA chest computerized tomography (CT) showed a 22-cm-sized huge breast mass with necrotic changes and multiple LN enlargements in the left axilla, both supraclavicular and parasternal areas including the left internal mammary and paracardiac LNs (Figure a). On 18 F-FDG positron emission tomography (PET)/CT, a huge uneven hypermetabolic mass was seen in the left breast, and there was no evidence of distant organ metastasis except for extensive hypermetabolic LN metastasis (Figure b).\nUnder strong clinical suspicion of inflammatory breast cancer, we performed a core needle biopsy on the tumor itself. Core-cut biopsy revealed a biphasic neoplasm with mild nuclear atypia and low mitotic activity, accompanied by leaf-like processes protruding into dilated ductal spaces, consistent with a low-grade phyllodes tumor. We discussed the discrepancy about the clinical diagnosis and core biopsy results at the preoperative multidisciplinary meeting that included an oncologist, a breast surgeon, a pathologist and a radiologist. We concluded the case was a carcinoma associated with huge phyllodes tumor and decided not to do further preoperative biopsy. The patient underwent a palliative modified radical mastectomy with level II axillary LN dissection. Macroscopic examination of the resection specimen revealed a firm and well-demarcated mass with hemorrhage and necrosis, measuring approximately 24 cm in the largest dimension (Figure a). This mass was composed of two separated tumorous lesions: phyllodes tumor and invasive carcinoma of no special type. Histologically, about 70% of the tumor area represented a classical phyllodes tumor showing variable benign to malignant histologic characteristics. The malignant phyllodes tumor was composed of highly cellular stromal cells, moderate and variable nuclear atypia, and increased mitotic activity (>10 mitotic figures per 10 high-power fields) (Figure b). In addition, in almost one-third of the tumor, frankly invasive carcinoma of no special type (Figure c) with foci of pleomorphic carcinoma was observed (Figure d). The skin was thickened and showed pathognomonic dermal lymphatic tumor emboli, consistent with inflammatory carcinoma (Figure a). The areas of margin between the two tumor lesions were evaluated by further re-cutting and serial section. On serial section, there was no histologic evidence of transition from phyllodes tumor to invasive ductal carcinoma, so the lesion was thought to be a collision tumor with invasive carcinoma of no special type and malignant PT, rather than ductal carcinoma arising from PT (Figure b). Metastatic ductal adenocarcinoma cells were detected in 16 out of 16 dissected axillary lymph nodes. On immunohistochemistry, both epithelial and stromal compartment of the PT as well as the carcinoma cells showed no immunoreactivity for estrogen and progesterone receptors and HER2, except for p53 overexpression on the carcinoma cells.\nThe wound healed completely without complication. Chemotherapy based on anthracyclines and taxanes was started and radiotherapy was planned. Six months after the operation, and after completion of eight cycle’s chemotherapy, the patient developed left supraclavicular and upper chest wall swelling, which was consistent with regional aggravation of metastatic carcinoma and was supported by image findings. She underwent a session of radiation therapy to the chest wall and neck, and platinum-based chemotherapy will be pursued in this patient as further treatment. She is doing well with good compliance to the chemotherapy and radiation therapy. |
A 12-year-old girl visited the plastic surgery department with a chief complaint of a growing mass on the dorsum of her nose. She had no history of nasal trauma, underlying disease, or congenital disease. The mass measured approximately 2.3×2.5 cm, had gradually grown over the past month, and was round and movable. The patient did not present with any noteworthy symptoms, including skin color changes or blockage of the nasal cavity, and the size of the nasal cavity on both sides was identical. She reported no discomfort other than slight pain associated with the growth of the mass and mild hypoesthesia on the nasal dorsum. We conducted two-point discrimination testing using a caliper to assess sensory impairment, and the result was 15–17 mm (reference: 10–12 mm) []. The patient complained of sensory abnormalities precisely in the area innervated by the infratrochlear nerve, leading to the suspicion of schwannoma in this area. Preoperative ultrasonography revealed a heterogeneous, echoic, solid cystic mass that did not invade the nasal septum, cartilage, mucosa, or bone. T2 magnetic resonance imaging (MRI) showed a round, well-defined, hyperintense mass. A direct incision was made because the mass was large; therefore, the open rhinoplasty approach would have been limited to the adhesion between the mass and soft tissue and securing the field of view. Under general anesthesia, an incision was made directly over the mass and resection was performed successfully. The round, encapsulated mass was completely removed. The patient had no significant complications and reported high cosmetic satisfaction. The diagnosis was confirmed by a definitive histopathological examination. The patient was monitored for 7 months after surgery, and an appropriate nasal contour was maintained. At a follow-up visit, we performed two-point discrimination testing around the mass. Sensation had improved relative to the preoperative findings, but had not returned to normal. There were no signs of recurrence or complications (). |
A 49-year-old super-super-morbidly obese female (BMI 90 kg/m²) with a past medical history of chronic obstructive pulmonary disease and cor pulmonale presented to the emergency department with altered mental status and difficulty ambulating secondary to lower extremity edema. Vitals revealed an elevated heart rate of 100 beats per minute and decreased respiratory rate of 10 breaths per minute. Physical exam was significant for lethargic mentation and denuded abdominal wounds under her pannus. Bi-level positive airway pressure and intravenous clindamycin were started due to hypercapnic respiratory failure and concerns for infection. Chest X-ray showed hyperinflated lung volumes without obvious signs of pneumonia (Figure ). Arterial blood gases demonstrated elevated serum bicarbonate, consistent with chronic carbon dioxide retention. The patient was admitted to the intensive care unit and emergently intubated due to worsening respiratory status. General surgery recommended discontinuation of clindamycin in favor of local wound care.\nOn hospitalization day seven, palliative care was consulted around goals of care and tracheostomy tube placement. Percutaneous endoscopic gastrostomy (PEG) tube or gastrostomy tube placement were discussed for nutrition; however, the ICU and surgical teams presented concerns of abdominal wall thickness and potential displacement into the peritoneal and abdominal cavity. The family was counseled regarding the risks and benefits and elected to proceed with a tracheostomy tube and PEG placement with full code status. Standard approaches for tracheostomy and PEG tube placement were performed. Directly post-procedure, however, the patient developed atrial fibrillation with a rapid ventricular response. Advanced cardiac life support (ACLS) protocols with phenylephrine, midazolam, cardioversion, and amiodarone were successively given in addition to a heparin drip, which stabilized the patient. Two hours after direct current cardioversion, the patient became hypotensive, and an arterial line was placed. After arterial line placement, the patient returned to atrial fibrillation and hypotension.\nFollowing substantial worsening in the patient’s clinical picture and vitals (oxygenation levels dropping below 86% and difficulty breathing), the critical and intensive care teams increased the positive end-expiratory pressure (PEEP) and tidal ventilation (VT) secondary to the patient's body habitus. During these attempts to stabilize the patient, it was observed that the balloon on the Shiley tracheostomy was deflated and subsequent attempts to inflate were unsuccessful. The patient was removed from the ventilator while manual ventilation took place and subsequently intubated orotracheally. Although the patient was able to be ventilated manually to 100% oxygen saturation after intubation and decannulation, however, would spontaneously desaturate despite increased oxygenation levels, ventilation rates, and vecuronium. Cardiopulmonary resuscitation (CPR) was initiated per ACLS protocol, in which return of spontaneous circulation (ROSC) was achieved for one minute, however, quickly became pulseless again.\nThe patient's family was notified during the start of the code and arrived during ongoing CPR. The family asked to stop CPR after 12 rounds of epinephrine. CPR was discontinued and the patient was asystolic. |
A 20-year-old man, presented to the emergency department (ED) with a history of difficulty in breathing through a tracheostomy tube (TT) since 2 days. Patient gave a history of a road traffic accident (RTA) 10 months ago, in which he sustained a head injury (diffuse axonal injury) which left him quadriplegic and bed-bound. He required prolonged hospitalization and was tracheostomized 7 days after his RTA to facilitate prolonged mechanical ventilation. He was weaned off mechanical ventilation and discharged home with a metallic TT. Tracheostomy care and suction was done by caregivers at home. He was regularly followed up at a hospital nearby where his TT was changed every month. Four months prior to present admission to our hospital, the patient had difficulty breathing and was taken to the nearby hospital, where his TT was changed. He was mechanically ventilated for 2 days and discharged once stable.\nThe present episode also began with difficulty breathing. The patient's caregiver tried to change the TT but failed. The patient turned blue and was rushed to the nearby hospital. The doctor there attempted changing the TT but could not, but he successfully intubated the trachea with a 5-mm ID endotracheal tube (ETT) and referred him to our hospital for further management.\nIn the ED, his heart rate (HR) was 120/min, regular and blood pressure (BP) 110/70 mmHg. He was breathing at a rate of about 40/min despite being ventilated with an Ambu bag. Air entry was decreased bilaterally and expiratory phase was prolonged. A suction catheter could be passed down the ETT but encountered resistance distally. An arterial blood gas (ABG) showed pH 7.12, pCO2 69, pO2 320, HCO3 38, and saturation 99%.\nHe was shifted to the ICU but the oxygen saturation by the time he was admitted to ICU had fallen to 84–86%. An urgent bronchoscopy was done through the ETT which showed a collapsible, bleeding, soft-tissue mass causing near-complete obstruction of the trachea, with only a small hole visible during air movement into and out of the lungs. The mass was vascular, bleeding to touch, and the bronchoscope could not be guided beyond the obstruction. 2–3 ml of adrenaline 1:200,000 solution was the sprayed through the bronchoscope which subsequently reduced the vascularity of the mass. Thereafter, the bronchoscope could be guided, though with some difficulty, through the visible hole. The growth was found to be about 1.5 cm long and beyond it the trachea was of normal calibre. The 5-mm ID ETT was pushed through the mass without significant bleeding. After this, the ventilation became considerably easier and the oxygen saturation picked up to about 94–96%. The patient was given dexamethasone 4 mg intravenous q6h for 24 h to reduce the edema. Computerized tomography (CT) scan confirmed luminal narrowing at the level of the sternoclavicular joint 4 cm from the carina with thickening of the posterior and right lateral walls of the trachea. Bilateral pneumothorax and extensive pneumomediastinum were also noted.\nThe granulation tissue was resected endoscopically the next day and the ETT was changed uneventfully to a distal-long stem TT to bypass the remaining obstruction. The patient was discharged next day in a satisfactory condition. |
A 16-year-old male subject visited the Department of Periodontics, The Oxford Dental College and Hospital, Bangalore complaining of bleeding gums following brushing and painless swelling in the left posterior area of the hard palate since 6 months. His medical and family history was noncontributory and was in good physical condition. The extraoral examination was unremarkable. No other abnormality was found.\nOral hygiene status of the subject was satisfactory. Intraoral examination showed the presence of swelling in the left posterior area of the hard palate of approximately 2 cm × 1 cm in size and color similar to that of palatal mucosa with slight bluish hue in the center of the lesion with a presence of sinus opening []. On palpation, the lesion was nontender and firm in consistency around the borders and soft at the center of the lesion. There was purulent discharge through the sinus tract.\nElectrical pulp vitality test results were positive. To track the sinus tract, intraoral periapical radiograph was taken with gutta-percha point placed through the sinus opening. The gutta-percha point extended till the apex of mesial root of a maxillary first molar. There were no signs of alveolar bone resorption in the panoramic, periapical, and occlusal radiographs. The floor of the maxillary sinus appeared intact. Complete hemogram showed all the parameters within the normal range. Computed tomography peripheral nerve sheath reported soft tissue density on the left side of palate causing smooth bony indentation [].\nInitially, the differential diagnosis of the swelling was made as periodontal abscess, palatal region vascular malformations such as hemangioma, mucocele, sialometaplasia, pleomorphic adenoma, adenoid cystic carcinoma, MEC, and adenosquamous carcinoma.\nIncisional biopsy was done to establish a definitive diagnosis, to assign a histological grade and prescribe adequate therapy. On histopathological examination, H and E staining showed three groups of cells, namely large mucous cells with an empty cytoplasm and peripherally placed nucleus, intermediate cells with basaloid nucleus, and epidermoid cells with eosinophilic cytoplasm suggestive of intermediate-grade MEC []. Mucicarmine stain, a specific stain for the diagnosis of MEC, highlights cytoplasmic mucin in the tumor cells. This staining showed large mucous cells with magenta pink colored cytoplasm indicating the presence of mucous, intermediate cells with basaloid nucleus, and lastly epidermoid cells with abundant cytoplasm. In areas, cystic areas filled with magenta pink colored material could be appreciated indicative of mucous, suggestive of intermediate-grade MEC []. The treatment consisted of wide surgical excision of the lesion with free surgical margins which was carried out under local anesthesia with an electrocautery. Boundaries of excision extended anteriorly until canine, medially up to midline, posteriorly until junction between hard and soft palate, and laterally until marginal gingiva. The Greater palatine vessel was ligated []. The excisional biopsy report confirmed the diagnosis of intermediate-grade MEC. Postoperative healing was uneventful.\nThe patient was recalled at 1, 3, 6 months and 1-year postoperatively to check for any recurrence. At 1-year healing was uneventful []. |
A 57-year-old right-handed female presented with a 4-year history of right leg weakness with equinovarus, and a reduction in grip strength in the right hand. Weakness was such that she had to lift her right leg in and out of her car with her hands. The following year, she began to experience numbness in the right hand as well as low back pain and urinary urgency. A course of intravenous methylprednisolone provided no benefit. Her condition slowly progressed but remained unilateral after 18 years, with no evidence of bulbar dysfunction. There have been no persistent sensory symptoms, though she has complained of cold extremities and acrocyanosis.\nThe patient was an ex-smoker. Her only past medical history of note was of curative (local) treatment for ductal breast carcinoma (11 years after onset of neurological symptoms). There was no family history of neurological disease.\nThe gait was spastic and hemiparetic, but ambulation was unaided. There was a pyramidal catch in the right upper limb and obvious spasticity in the right lower limb. Mild pyramidal weakness (Medical Research Council (MRC) grade 4) and hyperreflexia were noted in the right upper and lower limb.\nThere was an asymmetrical spastic paraparesis, worse on the right, and requiring a frame to ambulate. There were early flexion contractures of the fingers in the right hand with marked hypertonia in the right upper and lower limbs. Pyramidal weakness was noted in the right upper (MRC grade 4 proximally and grade 3 distally) and lower limb (MRC grade 3). Pathological hyperreflexia was now also evident in the left lower limb, but the left plantar response was flexor whereas the right was extensor.\nThe patient had begun using a wheelchair after fracturing the right radius and ulna in a fall, and had been catheterised due to impaired mobility. She had evolved significant amyotrophy in the right hand and forearm. |
A 17-year-old boy presented to the emergency unit complaining of retrosternal chest pain radiating to his left shoulder and back for one hour. Four days ago, he has started to receive penicillin treatment due to acute tonsillitis, and, on the fourth dose of penicillin, he had an episode of severe itching, nausea, and vomiting. It was accepted as penicillin allergy, and the treatment was changed to clindamycin therapy. One hour after the first dose of clindamycin, the retrosternal chest pain has started.\nHis electrocardiogram (ECG) on admission indicated acute anterolateral MI with ST segment elevations on leads DI, Avl, and V2 to V6 and also reciprocal ST depressions on leads DIII and aVF (). He had no risk factors for coronary artery disease, and his physical examination was unremarkable, but his blood tests showed elevated troponin levels as 1.4 ng/mL (normal range 0.00–0.11 ng/mL) and leucocytosis and eosinophilia. His echocardiography showed no pathologic signs of pericarditis, but he had slightly hypokinetic mid and apical anterior segments of left ventricle.\nHe was taken to the invasive laboratory, and coronary angiography was performed. His angiogram resulted as totally normal coronary arteries except at about 15 mm in length myocardial bridging at the mid portion of the LAD (). No further interventions were performed, and he was taken to intensive care unit. Combination of beta-blocker, acetyl salicylic acid and prednisolone therapy was started. His symptoms dissolved in half an hour, and during followup no symptoms repeated. His troponin levels elevated up to 18 ng/mL as peak and started to decrease then after gradually. His ECG findings resolved in the following days as the ST elevations and reciprocal ST depressions returned to baseline and the biphasic T waves observed as expected (). He was discharged on the fifth day, and, at the 3- and 6- month visits, he was free of cardiac complaints. |
A 17-year-old female presented with new onset right sided focal seizures that started on the day of presentation. She had three episodes prior to being seen at our casualty, each lasting about 5-10 minutes and was associated with confusion. At the casualty, she had a right sided tonic-clonic seizure that involved the face, upper and lower limbs. Four days prior to presentation, the patient started having a global headache that was associated with photophobia and painful eyes. She denied any fever, nausea, vomiting or a recent head trauma. Two weeks before, she had a tympanoplasty on the right ear for chronic suppurative otitis media without any immediate post-procedure complications. Her past medical history revealed a left ear tympanoplasty in 2011. She is HIV positive, acquired from her mother, and she has been on a combination of zidovudine, lamivudine and nevirapine since the age of 5 years. Her last CD4 cell count was 198cell/µL. Her mother died of HIV/AIDS when she was about 4 years old leaving her alone with her busy father. This has made her vulnerable to sexual abuses and she has had about eight reported episodes of rape since the age of 12 years. As a result, she was put on oral contraceptives (OCPs) since the age 13 years and was switched to injectable contraceptive Depo-Provera 4 months ago. She denied any history of alcohol intake, cigarette smoking or using illicit drugs. There was no family history of strokes, sudden deaths or clotting disorders.\nExamination after convulsion revealed an afebrile and anxious patient with a regular pulse rate of 127 beats/minute, a respiratory rate of 28/minute and a blood pressure of 131/43 mmHg. She had no lymphadenopathy and there were no bleeding or discharge from both ears. She had nuchal rigidity but without any neurological deficits. Examination findings of the abdomen, cardiovascular and respiratory systems were unremarkable. The patient's initial investigations revealed normal full blood count, renal and liver function tests. Her random blood glucose was 4.9mmol/L and she had negative antinuclear antibody test results. Cerebrovascular fluid examination revealed a slightly turbid fluid with 6 white cells/mm3 and 75 red blood cells/mm3. A computed tomography of the brain with contrast showed diffuse meningeal enhancement, white matter oedema of the left brain hemisphere and a filling defect at the confluence of the transverse cerebral veins suggestive of thrombosis of the sagittal vein. The patient was admitted for anticonvulsants and anticoagulation on the wards.\nOne day after admission she had about 10 episodes of sided convulsions, with residual right sided hemiparesis. The power of all the muscle groups in the right upper and lower limbs was grade 2/5. A Magnetic Resonance venography established superior sagittal and left transverse sinuses thrombosis (). Furthermore, there were venous infarction with haemorrhagic component and oedema in the left frontal, temporal, occipital and parietal lobes. In addition to continuation of anticoagulation, anticonvulsants were optimized to control convulsions. There was a reduction of seizure episodes over the subsequent five days and the patient's neurological deficit slowly normalized inthe next three weeks. She was then discharged home with mild hemiparesis and her subsequent outpatient visits showed no neurologic deterioration. |
A 51-year-old male weighing 131 kilograms (kg) presented to the emergency department (ED) via ambulance with altered mental status and slurred speech after undergoing cervical epidural injection with two milliliters (mL) of 2% lidocaine (40 milligrams [mg]) under fluoroscopic guidance in an ambulatory setting. He became unresponsive during the injection with subsequent brief convulsive activity for which he was given 2 mg of midazolam. Emergency medical services was called and found him obtunded with shallow breathing and low oxygen saturations requiring ventilation assistance. On arrival to the ED his breathing was spontaneous and erratic with low oxygen saturations. He remained somnolent with slurred speech, unable to answer questions appropriately or follow commands. Preparations were made for intubation given altered mental status and low oxygen saturations; however, oxygen saturations and mental status improved within the first 10 minutes of arrival and ultimately intubation was not required.\nOn cardiopulmonary monitor he was noted to have an irregularly irregular heart rhythm. Electrocardiogram showed atrial fibrillation with a rate of 82 beats per minute. Hemodynamically he was stable. He converted to normal sinus rhythm 20 minutes later. He reported no history of atrial fibrillation. Within 60 minutes of ED arrival the patient’s mental status was back to baseline without recollection of the events that had occurred after the start of the procedure. He only had chest wall pain, possibly from sternal rub or from any bystander chest compressions that may have been performed when he became unresponsive. Imaging studies obtained included the following: chest radiograph, computed tomography (CT) of the head, CT angiogram of the head and neck, and CT of the chest with intravenous contrast. No pertinent imaging abnormalities were identified. Serum/plasma levels of lidocaine and its primary active metabolite, monoethylglycinexylidide (MEGX) were obtained 15 minutes after patient arrival. Both levels returned undetectable. The patient was observed overnight in the hospital and remained asymptomatic and without any further dysrhythmia. He was discharged home the following day on aspirin 325 mg daily and with a referral to outpatient cardiology. |
A 68-year-old man presented with a 2-month history of a rapidly growing mass in the left neck on the thyroid gland region. He did not have any history of thyroid disease. He did not describe any symptoms like hoarseness, dyspnea, local pain, or weight loss. In laboratory evaluation, thyroid function tests, serum calcium, phosphorus, and parathormone were all evaluated as normal ranges. Serum calcitonin was measured as <2 pg/mL, which is low. Thyroid ultrasound revealed a 5 mm isoechogenic nodule on the right lobe and an 8 cm in diameter hypoechogenic, heterogeneous giant mass with irregular margins that was thought to be a nonthyroidal mass compressing the thyroid left lobe or may be a mass originating from thyroid gland and protruding outside the gland ().\nCervical lymph nodes had normal appearance. Tc-99m scintigraphy of thyroid gland was evaluated as a cold nodule occupying the middle and the superior regions of the left lobe ().\nAn ultrasound-guided fine-needle aspiration biopsy (FNAB) of the lesion was performed. It was reported as suspicious probable malign cytology such as medullary thyroid carcinoma. The magnetic resonance imaging (MRI) showed a huge amorphous, heterogeneous, expansive mass occupying the space from the left lobe of the thyroid gland to the retrotracheal area, which was 6 × 8.5 × 8 cm in diameter with irregular margins from the thyroid gland ().\nThe patient underwent surgery for exploration and tumor excision. Macroscopically, the tumor was well circumscribed and externally compressing the left thyroid lobe. As the mass was evaluated as distinct lesion from the thyroid gland intraoperatively, left thyroid lobectomy was not performed. Histology showed the tumor composed of spindle cells with patternless architecture and extensive stromal hyalinization ().\nThe lesion had a strong positive immunohistochemical reaction for CD34 in spindle cells (), stroma (), and diffuse bcl-2 staining ().\nThe final pathological diagnosis was solitary fibrous tumor. There was no complication postoperatively. Nine months after resection, the patient was free of disease. Neck region was evaluated by ultrasound; the left lobe of thyroid was evaluated clearly. Postoperative thyroid scintigraphy showed increased focal uptake in the middle and superior poles of the left thyroid lobe (). |
A 73-year-old female was transferred to our hospital with a history of fugacious amaurosis and transient sensory disturbance of the left upper limb, but was symptom free at admission. Magnetic resonance imaging showed an acute spotty cerebral infarction lateral to the caudate head, and magnetic resonance angiography revealed moderate stenosis in the right ICA (North American Symptomatic Carotid Endarterectomy Trial [NASCET], 58%). The right ICA was noted to be tortious, following a curved course medially. Plaque was present and markedly calcified. Also, the origin of the right ophthalmic artery was stenotic and had delayed blood flow on assessment by digital subtraction angiography (DSA) [].\nBased on the presentation, we performed CEA 21 days after onset to reduce the risk of recurrence. During surgery, no kinking stenosis was observed and video angiography with indocyanine green showed no delay in ICA blood flow. Postoperatively, clopidogrel 75 mg daily was started. Routine DSA revealed severe kinking stenosis of the right ICA. Ophthalmic symptoms appeared 20 days postoperatively. Fundus copy revealed cotton-wool spots in the right eye. Therefore, supplemental aspirin was added at a dose of 100 mg daily. Despite dual therapy, however, her symptoms recurred frequently and were refractory to further medical treatment. Moreover, no improvement of the stenotic lesion was seen on DSA or carotid Doppler ultrasound (peak systolic velocity [PSV], 226 cm/s) after 3 months. Therefore, CAS was performed under filter protection.\nDuring the CAS procedure, the kinking lesion was easily crossed with a micro guidewire and an open cell stent was deployed at the appropriate site. However, navigation of the stent delivery system beyond the kink was difficult. Postoperatively, both the ICA stenosis and the patient's symptoms improved. Fundus copy showed that there had been resolution of the cotton-wool spots in her right eye after CAS. No restenosis was seen by DSA performed 19 months after the procedure []. |
A 4-year-old female presented to our hospital with abdominal distension of 2 weeks duration. On examination, she had a right lumbar mass that was ballotable, normal blood pressure, and no dysmorphic feature. Computed tomographic (CT) scan of the abdomen showed an 11 × 8 cm mass arising from the upper pole of the right kidney []. The mass had both solid and cystic areas with calcification. There was a thrombus in the right renal vein extending along the inferior vena cava (IVC) till the right atrium. Her serum creatinine, urine routine examination, chest CT scan, and 24-h urine vanillylmandelic acid (VMA) levels were normal. Based on the clinical, radiological, and laboratory parameters a diagnosis of nonmetastatic Wilms’ tumor of the right kidney was made. At our center, we follow the SIOP-93 protocol for treating Wilms’ tumor.[] The protocol consists of 4 weeks of neoadjuvant chemotherapy with vincristine and actinomycin D followed by nephrectomy of the involved kidney and postoperative adjuvant chemotherapy and/or radiotherapy based on surgical and pathological findings. It is not mandatory to obtain tissue diagnosis prior to treatment in the SIOP-93 protocol if the clinical and radiological features are consistent with Wilms’ tumor; therefore, our patient did not undergo biopsy or fine-needle aspiration cytology (FNAC). She received 4 weeks of neoadjuvant chemotherapy; however, there was no clinical or radiological response to neoadjuvant chemotherapy. She was not considered suitable for surgical intervention due to involvement of the right atrium with tumor thrombus. The patient underwent a CT-guided biopsy of the right renal mass to ascertain the reason for poor response to neoadjuvant chemotherapy. The biopsy showed sheets of spindle-shaped tumor cells with moderate cytoplasm and hyperchromatic nuclei and immunohistochemistry (IHC) showed that the tumor cells were positive for vimentin, CD99, EMA, S100, and Bcl-2 and negative for CD10, desmin, and myogenin. The IHC was consistent with primary renal synovial sarcoma. No chemotherapy-related changes such as necrosis were seen in the pathological specimen. Based on the biopsy report of synovial sarcoma, the patient was treated with ifosfamide 1.8 g/m2 /day for 3 days and adriamycin 30 mg/m2 /day for 2 days; the chemotherapy was repeated every 21 days. After four cycles of chemotherapy, a CT scan of the abdomen showed a partial response with significant shrinkage of the renal mass and resolution of the right atrial thrombus. She underwent right radical nephrectomy with removal of IVC thrombus. The tumor and thrombus were completely excised. The postoperative histopathological examination was consistent with the findings of the preoperative CT-guided biopsy []. Polymerase chain reaction on the operative specimen was positive for SYT-SSX2 translocation consistent with synovial sarcoma. She received two more cycles of adjuvant ifosfamide and adriamycin. The patient relapsed 5 months after completing her treatment with extensive pulmonary and intracranial metastasis and died due to progressive disease. |
A 66-year-old woman was referred from the neurology department for bilateral tinnitus, hearing loss, and ear fullness that had progressed over the last two months. She also had headache, non-whirling type dizziness, left eyeball pain, arthralgia, and chronic cough. An otoscopic finding showed turbid ear drum in both ears. Four tone average of pure tone thresholds of the right ear were 43 dB (bone conduction) and 77 dB (air conduction), and those of the left ear 17 dB and 32 dB, respectively (, October 2008). Since follow-up audiometry after 2 weeks of treatment of antibiotics showed the disease to have worsened (, November 2008), myringotomy and a ventilation tube was inserted in the right ear. The ear drum and middle ear mucosa was markedly edematous. Spontaneous nystagmus under Frenzel glasses was absent, but an alternating bithermal binaural caloric test showed complete canal paresis of the right side. The direction of nystagmus was not changed with ice water irrigation from supine to prone position. Ophthalmologic examination showed no abnormality. Temporal bone computed tomography (CT) showed partial haziness in the middle ear and mastoid space and thickening of the tympanic membrane and middle ear mucosa. Brain magnetic resonance imaging (MRI) showed abnormal finding in pituitary fossa. It looks like mild pituitary gland enlargement or inflammatory haziness around pituitary fossa. Because of unilateral orbital pain and near the orbital apex lesion on MRI, neurologist suspected Tolosa-Hunt syndrome, and tried steroid therapy (prednisolone 60 mg per day for 25 days and then 30 mg per day for 15 days). During the steroid treatement, her right hearing improved (, December 2008). However, hearing loss was aggravated again (, February 2009) and left eyeball pain and cough relapsed about two months after steroid therapy. A chest CT was checked to evaluate cough, and it revealed multiple nodules in both lower lung fields and renal mass. Orbital CT revealed lacrimal gland enlargement. Biopsies of lacrimal gland, kidney and bronchial mucosa were preformed. Light microscopic examination showed dense lymphoplasma cell infiltration in the lacrimal gland, kidney and bronchial mucosa. Immunostaining for IgG4 showed strong immunoreactivity for IgG4 in infiltrating lymphocytes and plasma cells (). Serologic tests revealed elevated serum IgG of 19.20 g/L (normal range, 3.65 to 9.41 g/L) and elevated serum IgG4 of 2.45 g/L (normal range, 0.061 to 1.1214 g/L). Based on multiple organ involvement, elevated serum level of IgG4, and infiltration of IgG4 positive cells in the pathologic findings, we could diagnose IgG4 related systemic disease. We started immunosuppressive treatments consist of methylprednisolone at 60 mg per day and methotrexate at 12.5 mg per week, and her general condition and hearing markedly improved (, March 2009).\nUnder maintenance immunosuppressive therapy (prednisolone at 25 mg per day and methotrexate at 15 mg per week), there was further improvement of air-bone gap as well as bone conduction threshold (, May 2009). The dose of prednisolone was gradually tapered and she has been treated with 5 mg daily without further symptoms. We received Seoul National University Hospital Institutional Review Board approval for this study. |
An 82-year-old man presented to the emergency department with chief complaint of nonproductive cough, chest congestion, subjective fevers, hoarseness, and shortness of breath on ambulation for 2 days. He had chronic mild neck pain, which had been treated conservatively for the past 10 years. The patient had a history of 35 pack-years of smoking, which was discontinued 30 years prior to presentation. His medical history included hypertension, type II diabetes mellitus, and peripheral vascular disease with claudication, benign prostatic hypertrophy, colonic polyps, and multifocal osteoarthritis.\nOn examination, the patient had a grade 3/6 systolic murmur and decreased breath sounds at the bases bilaterally. Additionally the patient noted a 6-month history of unstable gait and loss of hand dexterity as well as bilateral upper extremity numbness and pain. Spine examination revealed decreased cervical range of motion. Neurologic examination revealed diffuse upper extremity weakness and hyperreflexia in the upper and lower extremities. He had negative Hoffman and Babinski signs. He then underwent an extensive metabolic workup, which revealed an elevated white blood cell count, elevated troponins, hyponatremia, and elevated blood glucose. Auxiliary tests included an abnormal electrocardiogram as well as elevated right hemidiaphragm with full inspiration on chest radiograph.\nThe patient was admitted to the hospital with persistent dyspnea. On hospital day 3, he underwent cardiac catheterization, which revealed extensive stenosis of the major coronary arteries, which was believed to be the main cause of his dyspnea. On hospital day 5, he underwent triple coronary artery bypass grafting. Despite the cardiac surgery, the patient's dyspnea did not improve. In addition, he developed new complaints of generalized weakness. He continued to complain of chest discomfort as well.\nMagnetic resonance imaging (MRI) and radiographic imaging of the cervical spine revealed extensive multilevel degenerative spondylosis with moderate to severe central canal narrowing from C2 to C7 and myelomalacia (\n). The patient underwent C2–C6 laminectomy and instrumented fusion with local autograft (\n). Lateral mass screws were utilized in the subaxial spine and pedicle screws were utilized at C2. Dome laminectomy was performed at C2. After surgery, the patient had gradual relief of dyspnea as well as improvement of strength. At 12-month follow-up, the patient was ambulating well and had improved neck pain. The dyspnea was completely resolved. His neck disability index had improved from 38% preoperatively to 30% at 1-year follow-up. The patient's chest X-rays showed that the right hemidiaphragm returned to its normal position. |
A 26-year-old Han Chinese woman presented with a lesion on her left medial tibial plateau. She was an office employee with no athletic history; she denied any injury or symptom before she experienced a sudden sharp pain and instability of the affected knee while going downstairs. Her symptoms did not spontaneously relieve after 2 days of rest so she presented to our hospital. A physical examination revealed a swollen knee joint with Floating patellar test (+), Anterior drawer test (−), Posterior drawer test (−), Lachman test (−), Abduction stress test (−), and Adduction stress test (−). A McMurray sign could not be done accurately due to pain and swelling in her knee from 20° of extension to 120° of flexion. She had no neurological symptoms and no other physical signs were apparent. Of interest, a mistaken diagnosis of a lower insertion avulsion fracture of anterior cruciate ligament could be made at first glance at X-ray and magnetic resonance imaging (MRI) (Figs. and ).\nShe was treated with arthroscopy (Fig. ). During the operation, a primary diagnosis of OCD on her medial tibial plateau was indicated by an osteochondral fragment near the lower insertion of her anterior cruciate ligament, and the fragment was removed arthroscopically. Microfracture of the defect area was then performed. Histological examination which was done by a pathologist after the operation supported the positive pathology of OCD.\nPostoperatively, weight-bearing was disallowed for the repair of the osteochondral defect where the microfracture was done, whereas quadriceps muscle training, such as straight leg raising, was encouraged. Continuous passive motion was also used for extension and flexion of her knee for 3 weeks after her operation. Three weeks later, rehabilitation began from partial weight-bearing up to total weight-bearing with no effusion of her knee joint. At 3 months of follow-up, she could walk and ride a bicycle freely except for a little pain after lengthy activity, particularly when going upstairs and downstairs. She was asked to avoid competitive activities for at least half a year or even for all her life, but muscle training was necessary; we suggested some helpful sports such as swimming. At 6 months, she was told that she could engage in long walks and even jogging with full knee flexion and extension. |
Case 1 was a 55-year-old man who suffered from alcoholic liver cirrhosis (LC) without other significant past medical or surgical histories. He was diagnosed with LC in 2001 and had 3 documented cases of spontaneous bacterial peritonitis (SBP). He underwent deceased donor liver transplantation due to uncontrolled ascites and hepatic encephalopathy in October, 2003. Fibrotic changes of the peritoneum were not observed during the operation. He underwent emergency operation for bleeding control after LT on postoperative day 3. His maintenance immunosuppressive agents were tacrolimus (FK), mycophenolic acid (MPA), and prednisone. His general condition continued to be stable. He was admitted for cholangitis 2 times in 2012 and treated with antibiotics without any interventions. He was admitted with abdominal pain, nausea, and vomiting 11 years after LT. His WBC count was within normal range (5.41 × 103/µL) and CRP level was increased (2.54 mg/dL). He complained of abdominal pain and distention, but rebound tenderness was not detected. The abdominal X-ray was not suggestive of mechanical obstruction. Contrast abdominal CT revealed a large amount of ascites and the small bowel wrapped within a fibrotic capsule (). His symptoms continued for 10 days after presentation and he, therefore, underwent surgical treatment. During the laparotomy, a thick fibrous membrane wrapping around the small intestine and colon was revealed. Careful dissection and excision of the thick membrane were performed to release the small intestine without additional injury to the intestinal serosa (). After surgery, tamoxifen 20 mg was administered twice a day and FK was replaced with mTOR inhibitor (sirolimus, target trough level: 5–10 ng/mL). Prednisolone was restarted at 4 mg twice a day. He was able to start oral feeding 5 days after surgery and was discharged 2 weeks later without any complication. The tamoxifen was maintained for 3 months. He was symptom-free for 1 year with continued treatment with sirolimus and predenisone. |
A previously healthy 34-year-old Korean female was admitted to a regional hospital for fever, myalgia and severe headache that had started 2 weeks ago. Brain MRI showed a high T2 signal intensity change and diffuse swelling of the right temporal lobe, insula and hippocampus (Fig. ). Lumbar puncture showed lympho-dominant pleocytosis. The patient was started on intravenous (iv) dexamethasone and acyclovir under a high suspicion of viral encephalitis.\nShe was transferred to our institution 4 days later due to persistent headache despite treatment. A noncontrast computed tomography (CT) brain scan taken at our institution showed a hemorrhagic transformation of the right temporal lobe, which was not observed on the initial MRI (Fig. ). Follow-up lumbar puncture showed 510 white cells per mm3 (82% lymphocytes), 144 mg/dL protein and 61 mg/dL glucose. CSF culture studies were negative for bacteria, fungi and tuberculosis. PCR of the CSF confirmed the presence of HSV1. The patient was free of neurological symptoms, with a Glasgow Coma Scale of E4M6V5, and was admitted for close observation and continuation of iv acyclovir. Corticosteroid treatment was discontinued upon her admission. On day 3 of hospitalization, the patient presented with a sudden onset of vomiting and severe headache. Brain CT showed an increased amount of temporal lobe hemorrhage and a leftward shift in the midline (Fig. ). Mannitol was administered but did not seem to have a significant effect. The patient became increasingly drowsy, and her right pupil became dilated. She underwent emergency right decompressive craniectomy, expansile duraplasty and ICP monitor insertion. Postoperative brain CT showed alleviation of midline shifting (Fig. ). The patient recovered fully 5 days after the surgery. Apart from mild intermittent headache and dizziness, she did not show any other significant clinical symptoms, including neuropsychological problems. There were no significant neurologic deficits upon neurological examinations performed by the attending neurosurgeon and neurologist. The patient was discharged after completion of 2 weeks of acyclovir and returned 2 months later for cranioplasty. She was followed up 3 more times after cranioplasty. She was stable, without any neuropsychological problems or neurologic deficits, and was able to successfully return to work as a public official. |
We report the correction of bilateral idiopathic Madelung deformity, in a 13-year-old girl, by callus distraction osteogenesis technique using the TSF external fixation system for the gradual restoration of the normal orientation of the radial articular surface and the simultaneous lengthening of the shortened radius.\nThis patient presented to our institution with distinctive the clinical features of Madelung deformity bilaterally with a volar translation of the carpus and hand and apex dorsal ulnar angulation of the distal radius (Figure ). In addition, the dorsally prominent distal ulna was obvious, due to its relative dorsal subluxation (Figure ), the estimated clinically 10° pronation of the distal end of the radius, and the restriction of wrist dorsiflexion and supination.\nAt the first clinical examination, there were distinctive radiological findings characteristic of a severe wrist deformity bilaterally, both on the P/A and lateral view (Figure ). The P/A wrist view demonstrated characteristic features such as premature fusion of the ulnar half of the radial distal physis, focal osteopenia in the ulnar portion of the distal radius, indicative of the occupation of the ulnar corner of the radius by the fibrocartilaginous Vickers ligament, the exostoses of the ulnar border of the distal radius, and carpal wedging, forcing the lunate gradually to move proximally at the apex of the wedge (Figure ). The lunate proximal subsidence (Figure ) was found to be 5 mm in both wrists (lunate subsidence is the vertical distance between the most proximal point of the lunate and a line perpendicular to the longitudinal axis of the ulna, passing through its articular surface). Increased ulnar tilting of the distal articular surface of the radius was found. The distal articular surface of the radius had an ulnar tilting of 38° in the left wrist (Figure ) and 36° in the right wrist (Figure ), with a normal range of tilting (21° to 23°). On the lateral wrist image, an increased palmar tilting of the distal articular surface of the radius bilaterally was found. The distal articular surface of the radius had a palmar tilting of 28° in the left wrist (Figure ) and 25° in the right wrist (Figure ), with a normal range of palmar tilting (10° to 15°). On MRI of both wrists, the Vickers ligament was clearly seen (Figure ). The calculated distraction lengthening of the distal radius needed for the restoration of a normal-oriented distal radial articular surface was 5 mm for both wrists that time. In addition, for the restoration of a normal-oriented distal radial articular surface in both wrists, the 10° pronation of the distal radius needed correction.\nThe surgical correction of the left wrist Madelung deformity was executed first because the left wrist Madelung deformity was clinically and radiologically worse than the right. The distal radial osteotomy was done as close as possible to the CORA deformity (Apex of the deformity), at the intersection of the proximal and distal anatomical axes of the radius. The ideal osteotomy was at the transverse bisector line (Figure ). The radial osteotomy (1 cm proximal to the open growth insufficient physis) was combined with Vickers ligament release at the time of the procedure.\nAt 1-year follow-up, the left wrist demonstrated good alignment of the forearm/hand relationship, with increased supination and dorsiflexion (Table ). The 1-year follow-up also showed worsening of the right wrist Madelung deformity, with its distal articular surface volar and ulnar tilting increased to 30° and 40°, respectively (Figure ). In addition, the lunate proximal subsidence was increased to 12 mm (Figure ). The calculated distraction lengthening of the distal radius needed for the restoration of a normal-oriented right distal radial articular surface was 12 mm. The surgical correction of the right wrist Madelung deformity with the use of the TSF was executed then. Complex limb deformity correction with external unilateral or circular Ilizarov ring fixators, using the callus distraction osteogenesis technique, is used in children but requires frequent, time-consuming frame modifications. The TSF system, allowing for the progressive, simultaneous correction of all the components of a multiplanar malformation of a limb, such as the Madelung deformity of the wrist, minimizes the required time of deformity correction. Furthermore, this hexapod system, assisted with a web-based software program, allowed the proper modifications of the prescription needed and more precise correction of the deformity. The TSF system was assembled with six fast FX struts and with two 105-mm full rings for the left wrist (Figure ) and with two 130-mm full rings for the right wrist (Figure ), selecting the proximal ring as the reference ring for the left wrist (Figures and ) and the distal ring as the reference ring for the right one (Figures and ). The reference ring was placed orthogonal to the reference fragment (proximal or distal radius). For the distal ring fixation, two 1.5-mm smooth wires were inserted to the distal radius (Figures and ). For the proximal ring fixation, two rancho cubes (Figure ) were used for the insertion of two 3.0-mm half-pins to the proximal left radius. On the contrary, one rancho cube was used for the insertion of one 3.0-mm half pin to the proximal right radius and one 1.5-mm smooth wire was inserted to the proximal right ulna (Figure ). The incorporation to the distal ring, bilaterally, of two 3.0-mm half pins, inserted to the second and fifth metacarpal, was necessary to stabilize the wrist joint and to prevent its collapse (Figures and ).\nPrecise A/P and lateral images are absolutely necessary to define the six deformity parameters (Figure ). The web software gave us the calculated minimum correction time of 26 days for the left wrist and 28 for the right wrist, and the schedule the patient was expected to follow for struts adjustments every day. This calculation was based on the six deformity measurements, the four settings of the reference ring, the initial six struts settings, the defined structures at risk, and the entered maximum safe distraction rate of 0.5 mm/d. The initiation of correction was started at the seventh postoperative day. The multiplanar deformity of the distal radius was corrected anatomically at the end of the scheduled prescription. The web-based planning program was adjusted twice until total deformity correction was achieved. The frame was removed, when callus formation was achieved (Figure ). A further immobilization of the wrist with a fiberglass cast for 3 weeks was required (Figure ) after frame removal. The 1-year follow-up for the right wrist and the 2-year follow-up for the left wrist showed a good aligned forearm/hand relation (Figure ) with increased wrist supination, radial deviation, and dorsiflexion (Table ), compared with the preoperative range of motion. |
A 4-day-old Asian Chinese female newborn was admitted to our department because she presented with six round, soft, multiple, pedunculated swelling masses with two on her upper anterior ridge and four on her lower anterior ridge. The size of the largest mass was 3.5×3cm (Figure ). No family history of hereditary diseases was reported. The baby was delivered in the 38th week of gestation via Cesarean. Her birth weight was 2650g. She was unable to close her mouth and thus feeding was not possible. Her respiration was normal. The size of the swelling masses increased slowly after birth. Because of the feeding problems, an immediate surgery was planned. General anesthesia was placed in spare to manage intraoperative complications including blood asphyxiation and other airway-related problems. The feeder vessels were seen to originate from the alveolar ridge. Hence, a transfixion suture was placed slightly away from the lesion on the alveolar ridge so as to achieve pre-excision hemostasis and minimize the chances of intraoperative bleeding, which could endanger the airway.\nAll lesions were well defined, firm, round, smooth and pink in color on the cut surface. All alveolar masses were excised surgically without complications under general anesthesia on the sixth day after the birth of the baby, who was discharged on the third postoperative day. Postoperative recovery and surgical site healing were satisfactory.\nThe excised masses were fixed in 10% neutral buffered formalin. The tissue was submitted for histopathological examination. Immunohistochemical analyses were also carried out using a panel of antibodies, including vimentin, Ki-67, smooth muscle actin (SMA), synuclein (Syn), neuron-specific enolase (NSE) and S-100. Corresponding positive and negative controls were performed in parallel for all the antibodies tested.\nMicroscopic examination showed a benign tumor composed of sheets of closely packed, large, rounded polygonal cells with abundant granular, eosinophilic cytoplasm and round to oval and lightly basophilic nuclei (Figure ). The overlying mucosa showed a well-differentiated, stratified squamous epithelium (Figure ). The tumor was stained diffusely but strongly for vimentin and NSE, and was focally but weakly positive for Ki-67 and negative for SMA, Syn and S-100 protein. The diagnostic hypothesis of congenital epulis of the newborn was confirmed based on both the histological details and immunohistochemical profile of the masses.\nFollow-up was conducted for the next 2 months of the baby’s life; no signs of recurrence were found (Figure ). |
A 59-year-old European man crashed his car into a concrete dam (Fig. ). Bystanders attending to the accident found him in cardiac arrest and started cardiopulmonary resuscitation (CPR) immediately. Sufficient CPR efforts were continued until the emergency services had arrived. The first recorded heart rhythm was ventricular fibrillation (VF). On inspection, no signs of injury were immediately visible and no skid marks were found. CPR was continued by physician-staffed emergency medical services (EMS) according to the current advanced life support (ALS) guidelines []. Return of spontaneous circulation (ROSC) was achieved after 30 minutes. He remained unconscious without any sign of muscular activity. He was intubated, mechanically ventilated, and treated with catecholamines during and post CPR.\nAlthough the car was severely damaged, the prehospital physician deemed a traumatic cause for out-of-hospital cardiac arrest (OHCA) unlikely. Based on findings indicative of myocardial ischemia in a post-ROSC electrocardiogram (ECG), acute coronary syndrome was suspected as the etiology of cardiac arrest. After telephone consultation with the trauma leader of the regional trauma center, the patient was transported to the trauma center with percutaneous coronary intervention (PCI)-capability primarily within 120 minutes of the accident.\nOn arrival at the trauma center, the patient appeared clinically stable. His heart rate was 65 per minute, systolic blood pressure was 150 mmHg, oxygen saturation measured by pulse oximetry was 94%, and body temperature was 34.2 °C. Signs of myocardial ischemia were found in the ECG (Fig. ). His pupils were found to be equal, round, and reactive to light.\nAfter primary evaluation in the emergency room a whole-body CT scan revealed findings listed in Table . An MRI scan (Fig. ) of his head and neck was obtained immediately due to the severity of the CT findings. Additional findings in the MRI scan are summarized in Table .\nThe medical and social history of our patient were provided by his family. Subjective overall health assessment found the married man, who was a father and grandfather, to be in good health. He had suffered a fall leading to a fractured scapula 8 years before this accident, which was treated non-operatively. Two years ago, he was assessed for suspected coronary heart disease by a specialist in cardiology, who could not substantiate this suspicion.\nHe was transferred to the intensive care unit (ICU) for further treatment. Halo fixation was installed because only ligamentous structures were disrupted in this case. This procedure is common and adequate in AOD when no cervical spine fractures are present [].\nDue to several episodes of severe bradycardia, transient transvenous pacing was conducted. Cardiac diagnostics showed an ischemic cardiomyopathy with recurrent episodes of ventricular tachycardia. Assessment via echocardiography was performed in the trauma room, 3 weeks and 2 months after the accident and revealed akinesia of the left anterior descending coronary artery (LAD) region and hypokinesia of the inferior wall after a suspected myocardial infarction and VF. Early coronary angiography could not be performed due to severe brain injuries.\nAlthough he was initially assessed to have a poor neurological prognosis from the perspective of the neurologists and neurosurgeons because of his severe brain injuries, he could be discharged from the ICU after 23 days; he was responding to verbal contact and was able to move all his extremities.\nAfter 23 days of treatment at the trauma center he was transferred to a hospital close to his home. Further in-patient treatment was continued by local protocol for further 33 days (timeline in Table ).\nHe was discharged to a neurological rehabilitation facility, where care and rehabilitation efforts were continued with great success. Three months after the incident the tracheostomy was surgically closed.\nCoronary angiography was performed 4 months after the primary event and revealed no coronary artery disease. Subsequently, he had to wear a life vest due to arrhythmia. He was defibrillated once by the LifeVest® 3 months after the trauma during his stay at the neurological rehabilitation facility. Finally, 6 months after wearing the life vest an implantable cardioverter-defibrillator (ICD) was installed.\nSix months after the trauma, he was fully conscious, spontaneously breathing, independent of help in everyday life, and mobile with walking crutches. However, he was unable to swallow granular feed due incomplete bilateral paresis of the hypoglossal nerve. His neurologic status is continuously improving; treating neurologists attested a high potential of restitution. |
A 60-year-old male patient with multivessel coronary artery disease, who suffered from a non-ST elevation acute coronary syndrome (NSTE-ACS) 1 month prior to admission, a nonsmoker with type-2 DM on metformin, peptic ulcer disease, and a history of alcohol abuse, was admitted to our institution for CABG. Just after the NSTE-ACS, a left ventricle (LV) thrombus was seen on one echocardiographic examination, but it was absent during follow-up. There was no deep venous thrombosis or bleeding diathesis history. On admission, the patient was on aspirin 75 mg (once daily) and enoxaparin 60 mg (once daily). Routine laboratory tests were within normal ranges (\n). There were no abnormalities on physical examination, apart from obesity (body mass index, 32.7 kg/m\n2\n) when the patient was admitted. The lower extremities appeared normal. There were no varicose veins, no signs or symptoms of venous insufficiency, and the past medical history was negative for both personal and family history of chronic venous insufficiency or varicose veins. The patient was operated on following the standard procedures. During LITA harvest, a cardiac surgery resident harvested the right SV using the open technique. The wall of the SV looked grossly normal. Upon dissection, the side branches were tied off and clipped, and a needle was placed at the distal end, while the proximal end was still not separated. An attempt was made to flush the vein with a solution containing blood (20 mL), heparin (15,000 IU), and normal saline (10 mL), while the distal end was closed with an atraumatic vascular clamp, and vein thrombosis was noted. Upon the separation of the distal end, a luminal thrombus was visible. The left SV was then taken down using the same protocol by an experienced staff cardiac surgeon, with the same result. Presence of a luminal thrombus was confirmed upon separation of the proximal end. Systemic heparin was administered, and normal LITA outflow was confirmed. Concerns regarding safety of cardiopulmonary bypass use were raised due to suspected thrombotic issue, and the approach was modified. The LITA–LAD anastomosis was completed off-pump on a beating heart.\nThe postoperative course was uneventful. On postoperative day 1, the patient received dual antiplatelet therapy with aspirin and clopidogrel and was discharged on day 6 with no signs of thrombosis or myocardial ischemia. Elective angioplasty of nongrafted vessels was scheduled, and a complete thrombophilia screening was done (\n). On the 3- and 12-month follow-up, the patient did well. |
We report a 27-year-old man who was referred to Imam Khomeini Hospital with chief complaints of pain and an intensive restriction in the range of motion (ROM) in the bilateral hip joints. He was diagnosed with HME 12 years ago while he had hip trauma, and multiple exostoses around the knee, ankle and shoulder joints were found in the obtained X rays ( and ). He was diagnosed as AS 3 years ago due to low back pain (LBP) accompanied with significant morning stiffness that lasted about 1.5 h together with laboratory and radiological findings. He was diagnosed as AS based on New York criteria including clinical criteria; a) low back pain more than 3 months improved by exercise and not relieved by rest, b) limitation of lumbar spine motion in sagittal and frontal planes and limitation of chest expansion, and radiologic criteria of bilateral sacroiliitis grade 2 or more ().\nThe patient’s sister and his 3 brothers have multiple exostoses without any family history of spondiloarthropathy or inflammatory arthritis. He has been taking sulfasalazine 1500 mg per day and indomethacin 75 mg twice a day because the bilateral hip joint ROM limitation had caused motion dysfunction. Flexion arc was limited to 30 degrees with a maximum flexion of 60 degrees. Motion was also restricted in the adduction-abduction arc as well as the rotator arc. Specific examination for AS including the Schober test and chest expansion was positive. Lateral bending was limited.The laboratory tests revealed mild anemia (Hb: 11 mg/dl, Hct: 37%, MCV: 79 fL) and a high ESR rate (93 mm/h). C-reactive protein and HLA-B27 were positive, but rheumatoid factor, ANA, viral markers and the Wright test were negative.\nMultiple exostoses were seen around the knee, ankle and shoulder joints in the X rays (). Both sacroiliac joints were fused completely and the joint spaces of both hips were narrowed (). A whole body bone scan showed increased activity in the metaphysis of multiple long bones that supported multiple exostoses (). Hip MRI revealed decreased joint distance and obliteration of both sacroiliac joints with subchondral bone changes due to sacroiliitis. Dueto severe bilateral restricted hip motion, the patient had impaired functional activity. Bilateral total arthroplasty was performed (). |
A 31-year-old man came to our emergency room seeking medical attention for severe neck pain. He informed us that 2 days before, after excessive consumption of alcohol, he had made an incision into the skin of his penis below the frenulum by means of a needle in order to insufflate air into the opening with a standard ball pump (fig. ) for autoerotic purposes. He had been suffering from severe pain in the neck area since the morning.\nBelow the frenulum, a reddened incision of approximately 5 mm in diameter could be seen which extended into the hypodermis. Apart from that, the penis was without pathological symptoms such as swelling, hyperthermia or redness. Also, the penis was not rigid and there were no crepitations. In the neck area and on the remaining trunk, crepitations could be felt. This was where the patient's pain was located, radiating dorsolaterally from the cervical area into the trapezius muscle.\nThe psychological exploration of the patient's sexuality clarified that he had distinctive sadomasochistic masturbation fantasies. The patient had not pursued these sadomasochistic thoughts so far because he had not been aware of them.\nThe laboratory results were unremarkable. Because the patient was capable of spontaneous and pain-free micturition and the external examination showed no anomalies, a cystoscopic examination of the urethra was not carried out. A CT examination of the abdomen revealed generalized subcutaneous emphysema extending from the scrotum to the abdomen and the cervix (fig. ).\nThe patient was admitted for observation. He received antibiotic treatment with a gyrase inhibitor in order to avoid an infection of the area around the frenulum. The medical focus was on the neck pain caused by the air gradually travelling through the hypodermis from the genital area to the cranial area. The pain was stabilized without difficulty by means of metamizole. After 2 days, it was no longer possible to detect any crepitations in the neck area. |
An 11-year-old male presented with a painless mass over the superior aspect of the globe for the last three months which was progressively increasing in size. The child had progressive, painless loss of vision in the same eye for the last three years. On ocular examination, visual acuity was no perception of light in the same eye and 6/6 in the other. Firm, non-tender mass measuring 4cm x 3cm was noted in the superior aspect of the globe (). It was covered by conjunctiva in the posterior half, while conjunctiva was adherent on the anterior half. Its surface was irregular with some vascularization. The cornea was hazy due to scarring, and inner details could not be evaluated. The examination of the left eye was within the normal limit. Systemic examination was normal with no palpable lymph nodes. B-scan ultrasound showed homogenous opacity in the anterior part of the eye with moderate reflectivity persisting up to 70dB. CT-Scan of the orbit revealed hyperdense focus attached to the anterior surface of the eyeball with surrounding thickening, likely foreign body with an irregular wall of the globe with areas of internal hyperdensity likely a hemorrhage or infection ().\nAfter consent, enucleation was performed with minimal manipulation under general anesthesia with the orbital implant and conformer. The specimen was sent for histopathological examination.\nGross examination of a cross-section of the enucleated globe revealed a solid grey lesion with exophytic and endophytic components (Figure 2.a and b). Histopathological examination revealed thick bands of polarized epithelium resembling medullary epithelium along with thinner cellular strands of acellular myxoid stroma (Figure 3.a). The tumor cells aggregated radially along the central space forming Flexner Wintersteiner rosette (Figure 3.b). It also involved the anterior chamber and vitreous cavity, but the optic nerve stump was free of tumor. This was highly suggestive of medulloepithelioma grade II. The origin of medulloepithelioma could not be confirmed.\nThe child is under regular follow-up. He is healthy, and there is no recurrence seen at one year of follow-up. |
A 46-year-old Bahraini female diagnosed as premature ovarian failure at the age of 29 years treated with hormonal replacement therapy presented with a history of epigastric abdominal pain and vomiting at the age of 37 years. Biochemical and radiological assessment showed features of acute pancreatitis in terms of elevated pancreatic enzyme level, and CT abdomen finding showed edematous pancreas with normal ductal system. It was attributed to hormonal replacement therapy after thorough investigation. Although the patient had stopped the implicated medications, she still had recurrent attacks of acute pancreatitis.\nSince there was no obvious cause found for her recurrent episodes of pancreatitis, autoimmune pancreatitis was suspected.\nThen, she underwent endoscopic ultrasound in 2015 which revealed mass swelling at the duodenal ampulla, and biopsy was taken. The biopsy showed ampullary adenoma with high-grade dysplasia (Figures and ).\nThen, the patient decided to go abroad for further assessment where she underwent Whipple's procedure and histopathology confirmed the presence of ampullary adenoma with high-grade dysplasia.\nUnfortunately, she continued to have recurrent episodes of pancreatitis despite the removal of the ampullary adenoma.\nIn 2016, while she was admitted under care of a surgical team for another episode of pancreatitis, she was reviewed by the rheumatology team to rule out autoimmune condition. Therefore, IgG4 level was tested (1.49 g/L (149 mg/dl)). The biopsy was reassessed and found to have increased IgG4-positive plasma cells around 30–40 per high-power field with the background of adenoma with high-grade dysplasia. Accordingly, she was diagnosed to have both IgG4-related disease and ampullary adenoma.\nShe was started on oral prednisolone 0.5 mg/kg and rituximab therapy with significant improvement over 1 year of follow-up as the pancreatitis attacks have reduced from around once in every month to around once in every 3 to 4 months after 3 months of rituximab therapy, and currently she remained attack free for around one year. |
We report the case of a 55-year-old Caucasian man with a 2 years’ history of multifocal HCC. Liver cancer developed in the context of a cirrhosis related to non-alcoholic steatohepatitis (NASH). At the time of HCC diagnosis, the patient was obese (body mass index (BMI), 36.9), affected by type 2 diabetes treated with oral hypoglycemic agents and by hypertension treated with sartans and diuretics. Blood tests were consistent with a compensated liver cirrhosis (Child-Pugh class A). Contrast-enhanced computed tomography and magnetic resonance revealed the presence of 10 HCC nodules (maximum diameter 2.8 cm) in the right lobe of the liver. He had received transarterial chemoembolization (TACE) with no response, probably because of the low vascularization of the lesions. Thus, treatment with sorafenib 400 mg twice daily had been started, with partial response visible in imaging tests performed during follow-up (no growth of known lesions nor appearance of new ones, while some nodules revealed the presence of necrotic areas). Because of the well-known high risk of hyperkeratotic HFS, the patient was instructed to generously apply emollients on hands and feet, and he developed only minimal skin changes without pain in the first 4 weeks of treatment. Two months after the start of sorafenib, he suddenly developed large painful nodules at inguinal folds, and such lesions progressively spread to trunk and axillae. The patient was treated with systemic antibiotic (amoxicillin 1 g twice daily for 12 days) and topical potent steroids, with no improvement. Because of the worsening of the clinical picture and the very intense pain reported by the patient, the dose of sorafenib was reduced to 400 mg daily, with maintenance of therapeutic efficacy, as confirmed by radiologic response, but no relief on skin toxicity. Physical examination showed multiple inflamed discharging nodules, localized on chest, axillae, lower abdomen, pubis, groin, and genital and gluteal regions (). Repeated bacteriological and mycological investigations revealed normal skin microbiota. Histologic examination showed dilated follicular infundibula filled with compact parakeratotic cornified cells and occasional vacuolization and dyskeratosis of the upper follicular epithelial cells. Dilated infundibula were often filled with neutrophils; cells were in some cases necrotic. A heavy infiltration of lymphocytes and plasma cells and, to a lesser extent, granulocytes (mainly neutrophils, with some eosinophils) surrounded follicular units; hyperplastic and dilated vessels were also visible (). Such protracted painful eruption severely affected the patient’s quality of life, especially because of the persistent purulent discharge, which interfered with daily activities. Treatment options were limited because of the presence of liver cirrhosis, concomitant illnesses, and related therapies. Systemic steroids were not recommended because of the presence of type 2 diabetes and hypertension, while their topical use was insufficient to control symptoms. Prolonged use of antibiotics was judged inappropriate because of negative microbiological tests. Oral isotretinoin was not considered in view of its potential liver toxicity. In agreement with hepatologists, in order to avoid treatment discontinuation, we decided to start anti-inflammatory photodynamic therapy (PDT) using aminolevulinic acid (ALA) as porphyrin precursor. After obtaining written informed consent, 10% ALA in polyethylene glycol ointment was applied in occlusion for 3 h on lesional and perilesional skin of pubis and chest; irradiation was then applied with diode red light at 630 nm. The light source was positioned at 50 mm from skin surface, thus achieving an irradiance of about 160 mW/cm2. The light exposure period was 8 min, resulting in a total light dose of 75 J/cm2. Fluorescence was detected using violet light at 405 nm and, after ALA application, was localized with high intensity in many inflammatory nodules. A visual analogue scale (VAS) was used to assess pain intensity. The patient was treated every 2 weeks for a total of four treatments. Severe reactions were referred after the first two sessions (mean VAS values, 9), with moderate discomfort recorded in the successive exposures (mean VAS values, 6). The treatment did not prevent the onset of new lesions, but determined progressive improvement of those already present, with marked reduction of purulent discharge and pain, ultimately resulting in improved quality of life (). Skin eruption quickly stopped after drug discontinuation because of liver transplantation. At 6-month follow-up, only hyperpigmented scars were observed. A written informed consent for patient information and images to be published in the study was provided by the patient. |
A 65- year-old male with past medical history of hypertension presented to the hospital with a six-month history of severe constipation intermittently. He had a bowel movement every two to three days. He had tried multiple medications and herbal treatment and at presentation required enemas to relieve his constipation. He, however, was passing flatus and had no other associated symptoms. He denied any abdominal pain, weight loss, change in stool or any significant family history. He reported a balanced diet. The patient was a laborer by profession.\nOn physical examination, at the time of presentation in the outpatient clinic, he did not show any signs of intestinal obstruction although reduced air entry was noticed bilaterally in the lower lung fields. His general physical examination and other systemic examinations were unremarkable.\nThe patient has advised baselines labs along with chest radiology and barium study. His baseline labs that included complete blood count turned to be normal. Chest radiography raised the suspicion of bowel in the pleural cavity. His barium study, however, confirmed the diagnosis as stated below (Figure ).\nThe patient was admitted for surgical closure of a hernia. An upper central abdominal incision was given and upon opening the peritoneal cavity, the defect in the diaphragm was clearly visualized (Figure ) along with the hernia sac and its contents, which were colon and greater omentum. The sac was opened and the contents were reduced back into the peritoneal cavity. Primary repair of the anterior defect in the diaphragm was carried out with interrupted ethibond 2/0 pledgeted sutures to the anterior abdominal wall, which was further reinforced with zero polypropylene suture.\nPostoperatively, the patient was mobilized, his diet progressed and was discharged once he was stable. He followed up in clinic after a few weeks and was doing well with regular bowel movements. Last follow-up after six months was unremarkable. |
A 16-year-old, healthy Caucasian boy, physically active and practicing sports, began suddenly complaining of a sharp pain in his left lower limb, which worsened while walking. The enlarged circumference, reddening and increased temperature of his left lower limb was observed as well. His history revealed that pain in his lower limbs and heels, marked fatigue and limited physical activity occurred as early as 2 weeks before the examination. Initially, he was hospitalized at the Department of Pediatric Surgery in a district hospital and deep vein thrombosis in his femoral vein up to approximately 10cm above the knee joint space was diagnosed. The results of laboratory tests revealed prolonged APTT: 64.7seconds. The remaining coagulation parameters including D-dimers were normal. The patient was administered low-molecular-weight (LMW) heparin (enoxaparin 40mg daily), diosmin, and compression therapy. Following discharge from the district hospital, he was admitted to the Department of Children Surgery and Oncology 12 days later, due to increasing lower limb edema and pain; femoral vein thrombosis in his left lower limb was confirmed. The results of laboratory tests showed markedly prolonged APTT, up to values that could not be determined by laboratory methods, and an increased level of C-reactive protein (Table \n). An intravenous antibiotic was applied (amoxicillin-clavulanate) and the dose of LMW heparin was increased (dalteparin 60mg daily). Compression therapy was continued. However, no clinical improvement was observed in his left lower limb after a month’s therapy; pain in his right foot occurred and his right foot was found to be colder. In addition, no improvement in his left lower limb was observed in a Doppler sonographic examination. On the right side, a two-phase blood flow with slightly weaker pulsation at the level of the adductor canal and the popliteal artery was found in his common femoral artery as well as in superficial and deep femoral arteries. Approximately 12cm to 13cm below the knee joint space, blood flow was growing weaker in his tibial arterial trunks and changed into one-phase flow, with medium pulsation. Peripherally, further deficiency of pulsation was noted in the posterior tibial artery (it was one-phase and characterized by low pulsation); no flow was detected in the right anterior tibial artery. Considering the course of the disease with venal and arterial thrombosis, a suggestion of an autoimmune disease was put forward. The patient was admitted to the Department of Pediatric Neurology 7 days later because of the occurrence of involuntary movements of his upper and lower left limbs as well as the left part of his face. Involuntary movements of the kind of hemilateral chorea involving his upper left limb were observed. A Doppler examination of his lower limbs was performed again and signs of deep vein thrombosis were found. The thrombotic process involved the femoral vein from the level of approximately 7cm below the peripheral ostium of the vena saphena magna (VSM), the popliteal vein and the proximal parts of the 3cm- to 5cm-long left lower limb veins. The lumen of the veins was totally filled with thrombi, which were 10mm to 11mm thick. The thrombi were partially organized. No signs of vessel recanalization were found. Thrombosis was not recognized in the superficial veins. The flow from the limb was via the patent VSM and the iliac vein (Figures \n and\n).\nBoth the deep and superficial venous vessels of his right lower limb were permeable and without parietal clots. The vessels were smooth and reacted properly to pressure. The flow in cervical vessels was assessed as well and it was found normal in the common internal and external carotid arteries. The echocardiography revealed normal structure and function of the heart. Due to persistent neurological symptoms computed tomography of the patient’s head was performed. Based on the results, thrombosis of the left sigmoid sinus was suspected.\nMagnetic resonance angiography was performed in order to verify the suspected lesions. However, the examination did not confirm thrombosis in the left sigmoid sinus. The suspicion of thrombosis of the internal carotid vein at the level of the skull base was put forward.\nThe boy was admitted to the Department of Pediatric Cardiology and Rheumatology for the purpose of performing further diagnostic studies. Analysis of the previous diagnostic findings was made and the diagnostic process was broadened according to the suspicion of a systemic inflammatory connective tissue disease.\nIn order to exclude a systemic connective tissue disease including systemic lupus erythematosus, a number of immunological tests were performed (rheumatoid factor, lupus erythematosus cell test, antinuclear antibodies, anti-neutrophil cytoplasmic antibodies), however, the results were negative. In addition, while searching for the causes of thrombosis, C protein, S protein and homocysteine concentrations were determined and were found to be within the normal range. No mutation of the allele of the gene factor V 1691 G-A (V Leiden) was observed. The patient was GG homozygote (normal sequence). No mutation in the prothrombin gene allele was found either. In both tests his deoxyribonucleic acid was isolated using the commercial kit (CHEMAGEN). The G20210A mutation in the prothrombin gene was detected using the restriction fragment length polymorphism (RFLP) method with the HINDIII enzyme (Fermentas). The mutation in the factor V Leiden was determined using the RFLP method with MNLL enzyme (Fermentas). Because of the fact that thrombosis occurring in the course of the APS was suspected, appropriate diagnostic procedures were performed. The obtained results are presented in Table \n.\nMarkedly increased levels of aCL and positive results for the anti-β2-GPI were noted twice. The patient did not present any sign, except chorea, suggestive of lupus erythematosus or any other systemic inflammatory connective tissue disease. Despite negative LAC, pAPS was diagnosed. He was treated with LMW heparin (dalteparin 15,000IU daily), acetylsalicylic acid at a dose of 75mg daily and risperidone, according to the neurologist’s suggestion. After 9 months of therapy, he remained in a good general condition, and no signs of chorea were observed. However, he complained of insomnia, troubles with memorizing and low mental concentration. Recanalization at the level of the left subpopliteal vein was observed, however, the superficial femoral vein from the level of approximately 6cm of the saphenous vein orifice to the popliteal vein was still filled with clots and no flow was observed. Similarly, a hyperechogenic thrombus was visualized in the distal part of the internal left carotid vein.\nHe was admitted to the hospital 6 months later due to painful testis edema. Ultrasonography confirmed another episode of venous thrombosis. The treatment was modified and LMW heparin was replaced with warfarin at the alternated dose of 5mg and 7.5mg every second day under international normalized ratio control. After 1.5 years of follow up the therapy with warfarin continues and he has no clinical symptoms. |
A 19-year-old male judo athlete presented with right knee pain that had persisted for three months. A valgus stress injury to the right knee sustained two years previously had been conservatively treated. However, the symptoms recurred along with several episodes of clicking and locking of the right knee during judo practice. A physical examination of the right knee showed a full range of motion with no effusion. McMurray's test was positive with lateral joint line tenderness. The knee was stable during the Lachman, anterior and posterior drawer, and medial and lateral stress tests.\nPlain radiographic findings of the right knee were normal. T2-weighted coronal magnetic resonance imaging (MRI) of the right knee revealed a small triangular fragment with a smooth edge over the normal lateral meniscus (). High-intensity signals were not evident within the normal lateral meniscus.\nArthroscopic examination revealed an upper accessory meniscus with a smooth, glossy surface overlying the lower normal meniscus (Figures and ). The upper accessory meniscus extended from the posterior horn to the middle segment of the lower normal meniscus and was firmly connected with the posterior horn and middle segment of the lower normal meniscus (). The upper accessory meniscus was attached to the capsule but was significantly thinner and more mobile by probing than the lower normal meniscus. In addition, the lateral meniscus had a meniscocapsular separation at the overlapping middle segment and was unstable by probing (). The medial meniscus, ligament, and articular cartilage were intact. We resected the upper accessory meniscus and repaired the meniscocapsular separation at the middle segment of the lateral meniscus using the FAST-FIX Meniscal Repair Suture System (Smith & Nephew, Andover, MA, USA) ().\nThe knee was postoperatively immobilized in a hinged knee brace locked in extension without weight-bearing for three weeks. Thereafter, partial weight-bearing and a 0°–90° range of motion of the brace were allowed. The full range of motion and weight-bearing were permitted at six weeks postoperatively. Running was allowed at four months and activity was unrestricted at six months. The patient was free of symptoms at 12 months after surgery and returned to his preinjury level of sport. At the final follow-up, plain radiography of the right knee joint showed no evidence of degenerative change and joint space narrowing. |
A 55-year-old man, a known hypertensive and asthmatic on therapy, presented with a three-month history of progressively increasing abdominal distension associated with dull pain in the abdomen. There was accompanying anorexia and loss of weight. He experienced mild breathlessness while sitting due to the abdominal distension. There was no fever, jaundice, features of gastrointestinal bleed or altered sensorium. Ascitic fluid cytology (at private laboratories) was reported as positive for Mycobacterium tuberculosis on polymerase chain reaction and the patient was started on antitubercular therapy. However, he had deterioration of liver function tests and the therapy was discontinued.\nOn examination, he had moderate ascites and mild pedal edema. Routine investigations revealed mild elevation of blood urea to 96 mg/dl and serum creatinine to 1.2 mg/dl. Biochemical and cytological analysis of the ascitic fluid showed it to be exudative in nature (protein 4.7 g/dl, total cell count 140/cu.mm.). Cytospin smears prepared from ascitic fluid showed lymphocytes and mesothelial cells in a mildly hemorrhagic background. In addition, occasional clusters and papillary fragments of cells having moderate amount of cytoplasm and central vesicular nucleus with distinct nucleoli were found [Figure –]. Focal acinar arrangement was noted. The clusters had a smooth outer border. The observed cell clusters resembled mesothelial cells, however ruffled cytoplasmic borders and intercellular “windows” were not identified in these clusters.\nFurther history was elicited, which revealed that the patient had undergone right radical nephrectomy two years earlier. Pathological examination of the right kidney showed a large 10 ×6 × 4 cm tumor with features of papillary RCC, type I [] confined to the renal capsule without extension to perinephric fat, hilar vessels or ureter. A review of the histologic sections of the renal tumor showed similar features in the cell clusters observed in ascitic fluid smears, and thus, a cytological diagnosis of malignant peritoneal effusion with cells from a RCC was made. Radiologic investigations (ultrasonography and CT scan) did not reveal any metastatic deposit in liver, left kidney or peritoneum. There was ill-defined thickening of the omentum beneath anterior abdominal wall. |
A 65-year-old male patient presented with a progressively increasing painless ulcer on the right side of the waist with a 6-month duration. Patient gave history of tightly tying a cotton dhoti over the waistline for the last 40 years. On local examination, there was a hypopigmented skin patch with scaling and ulceroproliferative growth of 7 × 5 cm with everted edges (Fig. ). There was no significant inguinal lymphadenopathy noted clinically and confirmed by FNAC. Changes of hypopigmented skin without ulceration were also observed on the left waistline. Based on the clinical suspicion, patient underwent wide local excision with clearance margin of 2 cm and primary closure of skin defect by mobilizing the skin flap. As regional lymph nodes were clinically and cytologically negative, they were kept under clinical surveillance.\nHistopathology reports confirmed the presence of squamous cell carcinoma with tumor-free margin (Fig. ). Postoperatively, the wound healed well without any complication, and the patient was discharged with a 3-month follow-up regime. After 1 year, locoregional recurrence was not observed on clinical evaluation.\nThe term dhoti cancer was coined in 1945 by Khanolkar and Suryabai [] which is a form of a waistline squamous cell carcinoma associated with wearing of dhoti by traditional Indian males. Similarly, wearing of saree by Indian women causing squamous cell carcinoma of the waistline was termed as “saree cancer” []. The data on this poorly understood squamous cell carcinoma of the skin is sparse in literature and hence the need for reporting this case with literature review.\nThe exact causation of dhoti cancer is ill understood, and there are many hypothesis put forward to explain its probable mode of causation. Constant friction at the site of tying of dhoti or saree causes friction of the waistline which is associated with dermatoses []. Other hypothesis for development of this waistline skin malignancy is continuous irritation which increases potential of malignant degeneration [] which could also be due to altered cycle of damage, irritation, and repair leading to malignant transformation [].\nAnother hypothesis put forward is depressed immunological state produced by the surrounding scar tissue [] and absence of lymphatic drainage from a scar which causes a significant delay in the host immunologic recognition and the antitumor immunologic response []. Squamous cell carcinoma developing in the scar was described by Marjolin in 1828 [], and saree and dhoti cancer could be a variant of scar cancer as constant irritation by dhoti produces depigmentation, glazing of the skin, acanthosis, scar formation, and later on malignant transformation that occurs in the scar. The dermal changes lead to scarring, and development of malignancy in this area of friction with scarring is the reason to call it as Marjolin-like cancer. A genetic hypothesis implicating human leukocyte antigen (HLA) DRG and mutation in P53 or FAS genes is also proposed [] for malignancy in the scar [].\nThe age of presentation in the present case that was 65 years with more than 40 years of wearing dhoti is quite consistent with its occurrence in the age group after 50 years as reported in literature. This suggests a prolonged exposure of more than 40 years before malignant transformation []. The features of these skin cancers are similar to Marjolin’s ulcer, being slow growing and well differentiated and lymph node metastasis is rare and that too will occur if growth infiltrates the surrounding skin or underlying muscle when inguinal lymph nodes will get involved requiring wide excision of the skin with grafting and radiotherapy to inguinal lymph nodes along with chemotherapy []. The treatment of a well-localized dhoti cancer is wide local excision with excision of the skin with acanthosis-like changes and primary closure or split skin grafting to cover the raw area. Long-term survival is not documented in literature due to the rarity of the disease. Another important aspect is follow-up of other side lesion where a change of acanthosis was observed. There is a need for an awareness drive for medical health providers and common public regarding these malignancies and regular screening of the waist skin of chronic dhoti users for any malignant transformation. |
A 54-year-old male with a past ocular history of angioid streaks and peripapillary geographic atrophy of both eyes presented for the treatment of a juxtafoveal choroidal neovascular membrane (CNVM) in the left eye. The CNVM had developed six months prior to his presentation to the clinic. He had been treated by an outside retina surgeon with two injections of bevacizumab, an anti-vascular endothelial growth factor agent that is commonly used to combat CNVM. His past medical history was significant for obstructive sleep apnea, osteoarthritis, and benign prostatic hyperplasia. Visual acuity was 20/20 in the right eye and 20/200 in the left eye, and he had an afferent pupillary defect in the left eye. Dilated fundus exam demonstrated geographic atrophy and angioid streaks in both eyes, as well as a CNVM in the left eye. Macular optical coherence tomography demonstrated retinal atrophy in both eyes, loss of inner segment/outer segment junction in both eyes, and a small amount of intraretinal fluid in the left eye (Figure ). After discussing with the patient, the decision was made to keep him under observation for the long term.\nAfter two years of stability, the patient started to complain of glare and haloes, which correlated with his worsening cataract in the left eye. He underwent uncomplicated cataract extraction with intraocular lens implantation in the left eye. His visual acuity in the left eye improved to 20/40. Three months after cataract surgery, his vision worsened to 20/60 in the right eye and 20/80 in the left. He also started to complain of worsening scotomas in the left eye, decreased color vision, and intractable headaches. A month later, he developed new flashes of light and floaters in both eyes. Multiple dilated fundus exams demonstrated stability to his initial exam. Given his worsening symptoms, a full-field electroretinogram was performed, which demonstrated extinguished scotopic and photopic responses in both eyes, indicating photoreceptor damage. Over the next few months, his vision worsened to 20/400 in both eyes.\nA systemic investigation into the underlying cause of his CNVM and optic neuropathy was pursued. Laboratory testing demonstrated an elevated nicotine level of 24.7 ng/mL (reference interval: <2 ng/mL) and elevated cotinine level of 280.2 ng/mL (reference interval: <20 ng/mL). The remainder of a toxicology screen was negative. Vitamin B12, folate, thyroid-stimulating hormone, antinuclear antibody screen, complete blood count, and complete metabolic panel were all within normal limits (Table ). MRI of the brain and orbits did not demonstrate any abnormalities. During clinic visits, he was consistently noticed to be chewing tobacco; he admitted to over 40 years of frequent chewing-tobacco use. He reported that he chewed approximately 70 grams of snuff daily. His preferred brand contains 3.8 mg/g of nicotine, resulting in approximately 266 grams of daily nicotine exposure. He also reported heavy alcohol use, with two drinks per day during the weekdays, and two to four drinks of hard liquor daily over the weekend. Ultimately, he was diagnosed with toxic optic neuropathy and choroidal neovascularization due to excessive tobacco and alcohol use. He was counseled to decrease his use of toxic substances, and he is currently working with his primary care physician on tobacco and alcohol use reduction. |
This patient, a five-year-old African-American female, fell at school and developed severe left knee pain. Three days after falling, the patient's mother contacted her primary-care physician where she reported her daughter's severe left knee pain that prevented her from walking and had worsened since the initial injury. At this point, the patient's left knee was also warm and erythematous. She and her daughter were referred to her local emergency department where radiographs of the patient's left knee were conducted and were read as unremarkable with no signs of trauma (). The patient was, therefore, diagnosed with a left knee sprain and was treated conservatively with pain control (ice and NSAIDs) and NWB without further intervention.\nEleven days later, the patient was seen by her primary-care physician for her persistent limp and intermittent pain, although her swelling had resolved. Anteroposterior view of the pelvis and bilateral frog lateral radiographs were ordered to rule out any hip pathologies, which could also cause an antalgic gait. All films were read by an attending radiologist and were deemed unremarkable. Relying on these reads, the primary-care physician recommended the patient to continue supportive care (NSAIDs and ice for pain, nonweight bearing for left lower extremity) and to refer to an orthopedist if symptoms persisted.\nDespite these treatments, the patient's pain and limp persisted, and the patient then sought a referral to a general pediatric orthopedist for further work-up. Although the child's pain had slightly improved, her limp remained. She described her left knee pain as worse at night but responsive to acetaminophen and ice. She had never felt anything like this before prior to her initial fall. Both the patient and her mother denied fever, weight loss, fatigue, malaise, or recent infection. On physical exam, her left knee did not have a palpable mass, muscle atrophy, or edema, but was tender to palpation. Given concern for the persistent pain and antalgic gait, repeat radiographs of the left knee were also ordered by the orthopedist and showed aggressive poorly defined lytic lesion centered within the left distal femur metadiaphysis extending up proximally 6 cm superiorly from the metaphysis with associated interrupted superior periosteal involvement (). A radiologist reviewed these findings and agreed with the orthopedist that based on these radiographic findings, there was concern for a primary bone malignancy. Arthrocentesis of the left knee was performed to rule out a possible infectious etiology, which was negative. Acute-phase reactants including c-reactive protein, sedimentation rate, and leukocyte count were all within normal limits. Magnetic resonance imaging (MRI) and needle biopsy were ordered to establish diagnosis. Contrast-enhanced MRI examination showed a enhancing mass centered in the distal femoral metaphysis approximately 7 cm in craniocaudal dimension with aggressive periosteal involvement (). Needle biopsy of the left distal femur was performed at the site of the lesion, and pathology demonstrated cores of tumor tissue consisting of pleomorphic tumor cells with frequent mitoses, foci of necrosis, regions of chondroblastic differentiation, and rare small foci of malignant osteoid production, consistent with the diagnosis of osteosarcoma. Additional imaging, including chest computerized tomography (CT) and whole body Positron Emission Tomography (PET), was obtained for tumor staging and demonstrated no evidence of distant metastases.\nThe patient was then referred to an orthopedic surgeon to discuss treatment options including oncologic and operative interventions. Given the concern for pathological fracture and possible subsequent contamination of healthy tissue from this tumor, the patient was placed in a knee immobilizer to limit weight bearing (NWB LLE with walker). The patient and her parents were referred to an oncologist to determine a chemotherapy plan. A typical treatment plan of osteosarcoma involves surgical resection and six cycles of chemotherapy (two cycles preoperatively as neoadjuvant therapy and four cycles postoperatively) with high-dose methotrexate, doxorubicin/dexrazoxane, and cisplatin.\nOperative treatments were discussed between the family and the attending orthopaedic surgeon in great detail including potential risks and benefits, and the family chose to proceed with Van Nes Rotationplasty. Radical resection of the left distal femur 17 cm from the joint line with femur/tibia osteoplasty with internal fixation and Van Nes Rotationplasty of the left lower extremity and left sciatic neuroplasty was performed. The procedure lasted around eight hours long without complications. Estimated blood loss was 100 mL or less. The patient was transferred to the PICU for monitoring postoperatively. There were no wound complications postoperatively, and the patient received trimethoprim-sulfamethoxazole and cefepime for antibiotic prophylaxis. Nine days later, the patient was discharged with referrals to physical therapy and the appropriative postoperative wound care instructions.\nThe patient was treated with chemotherapy for five months postoperatively and responded well. Since finishing chemotherapy treatments, the patient has continued with physical therapy and has been fitted for a lower extremity prosthetic. Postoperative radiographs have demonstrated appropriate healing of the rotationplasty (). Six-month postoperative Positron Emission Tomography (PET) showed no evidence of local or distant metastatic disease. There was minimal uptake in the left lower extremity at the location of the rotationplasty. The procedure with the appropriate adjuvant chemotherapy was deemed successful. |
A 42-year-old man presented to our pancreas multidisciplinary clinic after a computed tomography (CT) scan (), prompted by a 2-month history of generalized bloating and epigastric discomfort, that demonstrated a 11.2 × 9.6 cm heterogeneous solid appearing mass in the tail of the pancreas. The irregular mass had several small peripheral calcifications and lobulated contours abutting the spleen, stomach, and splenic flexure of colon without any direct invasion. He underwent a distal pancreatectomy and splenectomy with splenic artery lymph node dissection. Intraoperatively the large soft lobular cystic mass at the pancreatic tail was locally contained without any obvious invasion of surrounding structures or gross metastasis. Histopathological assessment of the mass established it as a pT3pN0pMx SPNP (CD56pos nuclear β-cateninpos chromograninneg and synaptophysinneg). Margins were negative without any lymphovascular or perineural invasion. The patient was discharged home after an uneventful period of convalescence in the hospital.\nFour years later, he was referred back to our clinic after discovery of a biopsy-proven recurrence in the splenic fossa (). The bulk of the tumor was densely adherent to the splenic flexure and gastric fundus and was resected with wedge gastrectomy and partial colectomy. A 4 cm nodule of tumor adherent to the diaphragm as well as omentum was removed by dividing the omentum and stripping the superficial layer of diaphragm. The tumor was soft, extremely friable, and fractured with minimal manipulation. It remained densely adherent to the left diaphragm, left kidney, and left adrenal gland. Eventually, we were able to dissect down through the Gerota's fat and strip the anterior capsule of the kidney clean to dissect the tumor off the kidney and the adrenal gland. The other end of the mass remained adherent to the diaphragm and was removed along with a portion of the diaphragm.\nFinal pathology report confirmed the presence of recurrent metastatic SPNP in omentum, diaphragm, accessory spleen tissue, and the gastric fundus. The patient recovered well from his surgery and was discharged home. He underwent CT surveillance at 3-month intervals per his medical oncologist and his first three scans showed stable postoperative changes without any evidence of local recurrence or metastatic spread. However, his next scan showed enlarged retroperitoneal paraaortic nodes that were found to be fluorodeoxyglucose (FDG) avid. He was started on capecitabine with stable disease on recent repeat imaging in April 2018. |
A 53-year-old male presented with a gradual onset of left-sided hemiplegia and weight loss of 14 kg over a period of ~4 weeks. His past medical history included COPD, hypertension and chronic hyponatraemia. There was no history of immunocompromise however he was malnourished. Of note, there was no history of foreign travel, intravenous drug use or household pets however the patient had undergone four dental fillings in the preceding 12 months. Neurological examination indicated MRC grade 2/5 power throughout the left upper and lower limbs with increased tone. The patient was apyrexic with a CRP of 68 mg/L.\nA CT brain revealed a right frontal mass lesion with surrounding oedema which was concerning for malignancy when taken in conjunction with the weight loss. Investigations for a primary tumour were negative including a CT of the chest, abdomen and pelvis in addition to upper GI endoscopy. Tumour markers did not reveal any abnormality. Additional testing for immunocompromise including HIV was negative and no primary source of infection was identified. A subsequent MRI brain revealed a total of five ring-enhancing lesions with vasogenic oedema distributed throughout both cerebral hemispheres with the largest measuring >2 cm (Fig. ).\nCommencement of dexamethasone (8 mg twice-daily) initially resulted in a good clinical response. Image-guided excision of the right frontal abscess was undertaken and culture results identified Nocardia farcinica. Despite appropriate treatment with intravenous linezolid combined with imipenem and the subsequent addition of co-trimoxazole, repeat imaging demonstrated a continued increase in size of the remaining abscesses. Two further operations were performed over a 4-week period in addition to ongoing anti-microbial therapy. Resection of all abscesses was achieved through right frontal, right parietal, left parieto-occipital and left temporal craniotomies. Microbiological analysis identified Enterococcus faecium in a second abscess and vancomycin was added to the antibiotic regimen. Post-operative imaging indicated complete resection and appearances were monitored with regular imaging (Fig. ). A prolonged course of dexamethasone was required to maintain neurological function. This may have contributed to a wound dehiscence requiring an uneventful wash-out and repeat closure in theatre. Ongoing seizure activity was controlled with anticonvulsants.\nDue to fluctuating symptoms, the patient remained in hospital for the duration of intravenous antibiotics over a 4-month period. Oral ciprofloxacin (750 mg twice-daily) was commenced for one year on discharge in addition to a 2-week course of oral Linezolid (600 mg twice-daily). Despite early post-operative expressive dysphasia and slow resolution of left-sided weakness, there were no neurological deficits noted on discharge. Following in-patient rehabilitation, the patient was discharged to his own home. A follow-up MRI at 6 months from discharge has shown no evidence of disease recurrence. |
A 59-year-old Caucasian woman presented with severe headache, repeated vomiting, and instability of stance and gait to our Accident and Emergency (A&E) department. She had been experiencing these symptoms for three hours. The patient had long-standing poorly-controlled essential hypertension, for which she took oral atenolol. The family denied head trauma or the ingestion of other medications. She was drowsy and had a blood pressure of 210/130 mmHg and a pulse rate of 110 beats per minute. Her lab tests (which included a coagulation screen) were unremarkable but her emergency non-contrast brain computed tomography (CT) scan revealed right-sided acute deep cerebellar hematoma with mild surrounding edema; no ventricular dilatation developed (Figure ). She was managed as a case of primary spontaneous hypertensive intra-cerebral hemorrhage. During the following two weeks, she showed a favorable improvement and then she was discharged home on enalapril, metoprolol, hydrochlorothiazide, and simvastatin. Her blood pressure was 125/75 mmHg at that time. She was able to stand and walk with some assistance and her speech was normal. Two weeks later, the patient came in for a scheduled follow-up visit. She was conscious and her speech was normal; she could stand and walk alone, and her blood pressure was 110/85 mmHg.\nEight weeks later, the patient presented with drowsiness, slurring of speech, vomiting, and inability to sit and stand unaided for one hour to our A & E. Her blood pressure was 190/100 mmHg. Her routine blood tests were within their normal reference range. An emergency non-contrast brain CT scan showed left-sided acute deep cerebellar hematoma, a contralateral site to the first hematoma (Figure ). The patient was treated medically and improved gradually over a two week period. On discharge, her speech was scanning and her gait was wide-based and ataxic. She could stand and walk alone with minor assistance. Because of the lack of expertise in our radiology department, conventional cerebral angiography was not ordered; however, a brain magnetic resonance angiogram (MRA) was done two weeks later and the result was unremarkable. We assume that our patient's hemorrhages were hypertensive in etiology. |
A 63-year-old man was initially admitted with status epilepticus and was commenced on phenytoin.
Magnetic resonance imaging (MRI) brain [] revealed a right temporal lobe enhancing lesion extending into the frontal lobe with extensive surrounding edema causing significant mass effect with a likely radiological diagnosis of glioblastoma. After discussion at the multi-disciplinary meeting, he underwent craniotomy and right temporal lobectomy for debulking of the lesion with a satisfactory 48-h postoperative MRI [] and an uneventful postoperative recovery.
Histology [] revealed widespread infarction, with some sparing of cortical layer I. There was an abundance of foamy macrophages and neovascularization. In addition, there was a marked inflammatory infiltrate with lymphocytes (largely CD3-positive T-lymphocytes) distributed throughout the brain parenchyma, with some additional perivascular cuffs. In addition, but less extensively, there were regions that were more hypercellular with atypical astrocytes (some of which were gemistocytic) together with mitotic figures, vascular proliferation and an elevated Ki67 (Mib1) proliferation index. Immunohistochemistry showed expression of herpes simplex virus (HSV) antigens. Following this, HSV1 DNA was detected by polymerase chain reaction (PCR).
As the inflammatory features were florid and predominant, and in light of the HSV immunohistochemistry and PCR, a diagnosis of HSV encephalitis was made, albeit with the proviso that there may be a neoplastic component on the basis of the astrocyte atypia and evidence of elevated cell turnover. Treatment with acyclovir was eventually started 3 weeks after surgery due to the delay in confirmation of diagnosis.
2 months following his initial surgery, the patient became increasingly lethargic and confused. A repeat MRI [] revealed recurrence of his temporal lesion and in view of his equivocal diagnosis, diffusion and perfusion-weighted MRI and MR spectroscopy was performed which revealed features suggesting a highly aggressive tumor. Hence, he underwent re-do craniotomy and debulking of the lesion.
Histology [] from the second operation differed from the first specimen by |
A 39-year-old ethnic Han Taiwanese male was healthy until he visited the ophthalmological clinic at Chang Gung Memorial Hospital due to the sudden onset of horizontal diplopia for a week and stated that the ocular symptom was more prominent in the early morning. On ocular examination, mild ptosis of the right eye with exotropia of 25 prism diopters was detected using Krimsky method, and limitation of eye movements in all directions was identified by diplopia test. Importantly, adduction, supraduction, and infraduction of both eyes were found to be impaired []. Moreover, the left eye levator and bilateral pupils were unaffected. The remaining results of his ocular and physical examinations were unremarkable. The complete blood counts, prothrombin time, activated partial thromboplastin time, blood chemistry profile, including serum vitamin level, and thyroid function test were all within normal limits. Although an initial diagnosis of myasthenia gravis was suspected, cerebral magnetic resonance imaging was arranged 3 weeks after the onset of diplopia, and the result revealed a midbrain hemorrhage [circles, ]. Mono-ocular occlusion was arranged with improved symptoms, and the patient was referred to a neurologist for further management where edrophonium test showed negative result. Two months after the onset of diplopia, sudden onset dizziness accompanied by nuchal pain as well as bilateral leg muscle weakness were noted, and then, cerebral magnetic resonance angiography [circle, ] revealed a hemorrhage over the brainstem that was associated with mass effect and hydrocephalus, but no vascular anomaly was noted []. Then, the patient became bedridden, and the follow-up neurological examination identified headache insomnia, dysarthria, dysphagia, and decreased muscle power (Grade 4 at upper limbs and Grade 2 at lower limbs). Nine months after the onset of diplopia, the patient exhibited poor appetite, yellowish sputum, and general weakness after choking before being admitted to our internal medicine ward under the suspicion of aspiration pneumonia. The patient died as a result of aspiration pneumonia-induced respiratory failure within the same month. |
A 66-year-old Caucasian man presented to our hospital with a history of thyroid lymphoma treated with chemoradiation therapy (60 Gy). Subsequent surveillance revealed a new lesion within the thyroid gland, and workup demonstrated a localized papillary thyroid carcinoma treated with total thyroidectomy. Postoperatively, he developed left true vocal fold paralysis due to close adherence of the primary tumor to the nerve, and 4 months later he developed an acute episode of airway distress, presumably caused by laryngeal edema, that required an emergent tracheostomy. The laryngeal edema gradually resolved, and his tracheostomy tube was successfully downsized and he was decannulated. He was then noted to have both a small tracheoesophageal fistula as well as a large TCF. The tracheoesophageal fistula was successfully managed with a sternocleidomastoid muscle rotational flap. Bronchoscopy revealed a TCF that was 1.4 cm × 1.6 cm in size and had an adjacent 1.0-cm section of proximal tracheal stenosis (Fig. ). Given his history of radiation, failure of previous primary and rotational graft closure, and the size and complexity of the TCF, a decision was made to repair the defect using a three-stage approach.\nThe initial stage involved implanting a conchal cartilage graft with the concave portion facing superficially on the ulnar aspect of the distal left forearm. Two months later, owing to the patient’s dense hair distribution over the ulnar forearm, a second-stage procedure was performed to place a buccal mucosal graft over the conchal cartilage graft after removing the hair-bearing skin. After allowing 6 weeks for healing, the third stage was performed. This stage involved transferring the radial forearm free flap (RFFF) with cartilage and mucosal graft for closure of the TCF and augmentation of the associated tracheal stenosis (Fig. ).\nDuring this procedure, the conchal cartilage covered by mucosa was inset into the tracheal defect with interrupted and vertical mattress 2-0 polydioxanone sutures, followed by application of a layer of fibrin glue. Support was augmented with a 1.2-mm absorbable miniplate (LactoSorb; W. Lorenz Surgical, Jacksonville, FL, USA). Two of these plates were used to secure the free flap graft to a trachea (one superiorly and one inferiorly). A narrow strip of the distal flap was deepithelialized to allow for the remaining skin to be used in the external skin closure. Additional RFFF soft tissue was positioned between the tracheal closure and skin. The radial artery was anastomosed to the right superior thyroid artery, and the cephalic vein was anastomosed to the right external jugular vein. Penrose drains were placed in the neck (Fig. ).\nThe patient was extubated in the operating room following reconstruction. The patient was advised to avoid straining, speaking, and coughing in the immediate postoperative period. He had no issues with breathing or wound healing in the immediate postoperative period or at 6, 12, 18, and 60 months postoperatively. |
A-17-year-old previously healthy female presented to THP with a history of fever for 2 days associated with body aches and nausea. She didn’t have any abdominal pain, bleeding manifestations or postural symptoms. On examination, she was flushed and febrile but was not pale or icteric. She was mildly dehydrated. Blood pressure was 100/70 mmHg, pulse rate 100 beats/min and capillary refilling time (CRFT) was less than 2 s. On abdominal examination, there was no free fluid. Lung fields were clear on respiratory system examination. Other systems examination was normal.\nHer NS1 antigen was positive and serotype was identified as DEN1. She was managed as dengue fever with continuous monitoring. On the 3rd day of fever, she complained of retrosternal chest pain and undue tiredness. At that time her cardiovascular system examination was normal and electrocardiogram (ECG) showed acute T wave inversion in V2-V5 leads. Troponin I was negative and 2D echo showed global left ventricular hypokinesia and mild impairment of LV function. Ejection fraction was 40–45%. She was treated as having dengue fever complicated by myocarditis. Intravenous hydrocortisone 200 mg 8 hourly was administered for 2 days to reduce myocardial inflammation. On the 4rd day following admission, she complained of abdominal pain and ultrasound scanning revealed free fluid in hepato-renal pouch. Blood pressure was 100/70 mmHg, pulse rate 70 bpm, and CRFT was less than 2 s. She was taken to High Dependency Unit (HDU) and was managed as having DHF complicated with myocarditis with continuous monitoring and with careful administration of fluid to avoid fluid overload. She was discharged on day 7 of illness after recovering from critical phase of dengue fever. She was advised on limiting physical activities. During the follow up on day 14 of the illness, ECG showed reversal of T inversions. Echocardiogram showed improvement of left ventricular function with an ejection fraction of 55%. |
We present the case of a 3.5-year-old Iranian girl who was referred to us from pediatric surgery clinic because of a gluteal mass. Her parents informed us that the swelling of the right buttock of their daughter had been present since her birth. The size of the mass had been increasing gradually. They did not mention any bowel or bladder dysfunctions. A local physician had aspirated the cyst twice, 8 and 4 months prior to this presentation. Clear fluid had been aspirated each time, bur the mass would always reappear after several weeks. Unfortunately the results of the chemical analysis of the fluid were not available.\nA physical examination of our patient showed a soft, 3 × 4-cm fluctuant and non-pulsatile mass over her gluteal region. It was completely covered with normal skin without any vascular or hairy stigmata. The size of this mass did not increase with crying or coughing. Her transillumination test results were positive. Her midline was normal, with no sinus tract or swelling (Figure ).\nResults of her sensory, rectal tone and lower limbs motor exams were within normal limits. No abnormal skin lesions or skeletal deformities were found. The curvature of her spinal column appeared normal.\nResults of her routine laboratory investigations were also normal. An anteroposterior X-ray examination of her lumbosacral region showed spina bifida of the L5 and S1 vertebrae (Figure ).\nAn ultrasound scan showed a very well-defined purely cystic, oblong lesion measuring 3.7 × 4.5 × 2.2 cm in size in our patient's right upper gluteal region under the gluteal muscles. There was no evidence of internal echoes or solid component or septae within the sac. We planned to perform myelography but unfortunately found it impossible because of our patient's allergic sensitivity to the contrast agent (omnipaque). A lumosacral magnetic resonance imaging (MRI) showed a well-defined cystic mass measuring 36 × 21 × 45 mm in her right buttock. The cystic mass appeared as a low signal intensity area on T1-weighted images and a high signal intensity area on T2-weighted images, which was similar to her cerebrospinal fluid (CSF) signal (Figure ). No obvious communication between the cyst and the spinal canal was perceivable. The position and shape of the cord, roots and lying of the conus were unremarkable. The findings were suggestive of lateral meningocele.\nA voiding cystourethrography showed a normal voiding pattern of our patient's bladder and urethra. There was no evidence of vesicoureteral reflux.\nWe then decided to perform resective surgery. After performing a transverse incision at the equator of our patient's lesion, we found a cystic, relatively thin-walled mass with a smooth pink exterior layer and a shiny creamy interior surface lying under the right gluteal muscles. We aspirated the cyst and clear fluid came out of it (Figure ). Opening the cyst, we saw a very small aperture at the level of the ilium. We further extended the hole using microdrill and Kerrison punch along the tract, through which we saw communication between the cyst and the thecal sac through the spina bifida at the sacral canal. No neural tissue was found lying inside (Figure ). After performing a ligature of the aperture of the cyst, we resected the walls and we closed the wound in layers. Our patient's postoperative recovery was uneventful.\nOn histopathology, we found that the wall of the sac was composed of fibrous tissue that had a lining of flattened to cuboidal epithelium. Polymorphonuclear leukocytes, histiocytes, and giant cells were also noted (Figure ). The final pathological diagnosis was reported to be a meningocele with inflammatory reaction. Analysis of her intracystic fluid showed the following: red blood cells = 100/cc, white blood cells = 2500/cc (polymorphonuclear = 72%, lymphocyte = 28%), glucose = 60 mg/dl and protein = 45 mg/dl.\nOur patient was found asymptomatic when she was presented for follow-up examination after 18 months. |
A 36-year-old lady presented with redness and decreased vision in right eye for 6 months. She was earlier diagnosed of a cavitary lung lesion, presumed secondary to tuberculosis and treated with anti-tubercular treatment for 4 months. At presentation, vision was nil light perception in the affected right eye. The left eye was essentially normal. Examination revealed conjunctival congestion with a diffuse vascularized conjunctival mass in the inferotemporal bulbar conjunctiva and proptosis []. Iris neovascularization, 360° closed angles with intraocular pressure of 48 mm Hg, and a choroidal mass with total retinal detachment were noted. Ultrasound of right eye revealed diffuse choroidal thickening with uveal mass, widening of the tenon's space and diffuse orbital tumor []. Magnetic resonance imaging scan revealed uveal mass and diffuse orbital tumor []. A diagnostic needle biopsy of the uveal mass revealed the presence of atypical cells in necrotic background suggestive of malignancy. The patient underwent lid-sparing exenteration of the right eye []. Histopathological examination of the exenterated specimen revealed the presence of mucoepidermoid carcinoma with multiple tumor emboli within the blood vessels of the orbital tissue [Fig. and ]. Extensive invasion into the orbital tissue, muscle fibers, sclera, choroid and optic nerve was noted. Postoperative period was uneventful. Before orbital exenteration, she also underwent computed tomography-guided biopsy of the cavitary lesion in the left lung that revealed squamous cell carcinoma (SCC) [Fig. and ]. In view of the systemic spread, radiotherapy was advised. At 3 months follow-up, two cycles of radiotherapy were completed, and the right socket was healthy.\nMucoepidermoid carcinoma of the conjunctiva often presents as a limbal or bulbar conjunctival mass with symptoms of chronic irritation. It was reported for the first time in conjunctiva by Rao and Font in a series of five cases in 1976, all the five cases had recurrence within 6 months.[] Clinical morphology can range from simple conjunctival nodule to leukoplakic, infiltrative, ulcerative, and papillomatous lesions. Histopathology of MECC is variable. Our case revealed tumor cells arranged in lobules with fine septa separating them. The cells had squamoid morphology with duct-like structures and were seen infiltrating sclera and orbital tissue. Tumor emboli were seen in blood vessels. The tissue was positive for alcian blue staining [].\nReview of literature suggests the role of intraocular invasion by tumor cells to be implicated in the mucinous differentiation in the tumor, and perhaps mucinous metaplasia is due to intraocular factors.[] Lymph node involvement has been described, but distant metastasis is unusual if the tumor is treated aggressively.[] Seitz and Henke. suggested that early enucleation including normal appearing tissue next to the globe should be considered for treatment of primary mucoepidermoid carcinoma to avoid later exenteration or metastasis.[] Johnson et al. and associates reported two cases of MECC presenting with cervical lymph node metastases, in a series of 30 invasive secondary orbital SCC that were treated with exenteration and radical neck dissection.[] Recurrence is also a common feature of MCC occurring to the extent of 79%, most commonly in the first 6 months of follow-up.[]\nIn our patient, extensive orbital disease at the time of diagnosis necessitated exenteration and vascular invasion was seen histopathologically. There was a large cavitary lesion in the left upper lobe of lung with central necrotic area and nodular enhancing soft tissue toward the hilum with a fairly thin, uniform wall. Systemic metastasis was treated with radiation therapy. The patient was recovering well. Given the high frequency of misdiagnosis and incorrect management, it is essential that proper histopathological examination be carried out.[] The treatment strategy is decided once the status of intraocular spread as well as distant metastasis is known. |
We here describe the case of a four-year-old female child who suffered burn injuries while onboard a stationary aircraft on her way home after a holiday abroad. The airbag was concealed within her seat belt unexpectedly and inexplicably deployed before departure, resulting in burn injuries to the left side of the face, left jawline, left side of the chest, left upper limb, and left anterior thigh. Before departure, she was assessed by paramedics and deemed fit to undertake the eight-hour transatlantic flight to the United Kingdom.\nOn landing, she was referred to the local hospital emergency department where she was seen approximately 12 hours after the injury. She was in pain but was hemodynamically stable and had a patent airway. She was noted to have cutaneous injuries with associated swelling on the left side of her face and body but there was no obvious auditory, ophthalmic, dental, or internal organ trauma on clinical examination and no associated fractures on radiologic assessment. Her past medical history was unremarkable, with no regular medication or known drug allergies and she had normal developmental milestones. She was referred to a specialist center for a review of her cutaneous injuries, given oral morphine for her pain, and transferred across by ambulance.\nDetailed examination of her cutaneous injuries at the specialist center revealed a combination of blistering, friction-abrasion type injuries with bruising and swelling on the left malar area, cheek, and chin which were consistent with superficial partial-thickness burns. There was a strip of skin overlying the left jawline where the burn was slightly deeper and was clinically consistent with a partial thickness burn (Figure ). There was a small zone of contusion without epidermolysis over the left upper chest consistent with blunt trauma. Patchy non-circumferential superficial partial-thickness burns were also noted on the anterior and medial aspect of the left upper limb extending in a patchy distribution from the anterior left axilla down to the left wrist along the medial border of the limb. There were no associated fractures but the range of motion was limited at the left elbow and wrist secondary to pain. A patch of the friction-abrasion-type burn was also seen over her left anterior thigh. There were no concerns regarding inhalational injuries on clinical examination and a formal airway assessment was not warranted. The pH of the burn sites was normal but suspecting a combination etiology (thermal, chemical, and mechanical burns), the burn sites were cleaned with saline and the facial burns were treated with Vaseline and non-adherent silicone dressings were applied to the patchy burn on her left upper limb. The left upper limb was placed in a sling for comfort and she was admitted to the hospital for analgesia, observation, and to facilitate feeding. She also underwent an assessment by a pediatric clinical psychologist to help her and her parents deal with the shock of her accident and injuries. Her inpatient stay was unremarkable, there were no safeguarding concerns and the child was discharged after 48 hours later. She remained well and her occasional complaints of itching at the burn sites were responsive to oral antihistamines.\nA week after the injury at the first out-patient clinic review, the burns sites appeared to be healing with no evidence of infection. The non-adherent dressings applied to the sites on her left upper limb were replaced and the parents were advised to continue applying Vaseline to the face. The child had had some difficulty eating and drinking and the parents had encouraged oral intake through a straw which she had managed. Sleep disturbance, enuresis, clinginess, and fear of heat were reported on review by the pediatric clinical psychologist. Two weeks after the injury, at the second out-patient review, the burn sites had healed (Figure ). The child was referred to the scar management team who advised the use of moisturizer over the zone of injury and recommended sun protection.\nFive weeks after the injury, a third out-patient clinic review was undertaken as the child’s brought forward concerns regarding the “glowing red” appearance of the healed areas on the left side of the child’s face and jawline when the child was physically active or was flustered. On examination, no hypertrophy of the healed areas was observed. The parents were reassured that the scar was in its remodeling phase and may appear red in warm temperatures or appear dusky in cold weather for up to 12-24 months after the injury (Figure ). The need for regular application of a non-perfumed moisturizer and the importance of sun protection was reinforced. On subsequent visits, the complaint was still there, flushing in warm weather and mild mottling (“blemish”) in cold weather. The senior author referred to this phenomenon as “intermittent dermal hypervascularity” and felt that it would settle in 18-24 months, which it did.\nThe child remained fearful of aircraft despite having traveled on them after the injury. She also suffered nightmares for months and slept in with her mum and dad for the first eight months and after that preferred to sleep with her younger brother. Her parents explained that she had been so severely emotionally affected by the accident that she continued to talk about the incident daily for a few years after the accident. In this regard, the early involvement and follow-up by a pediatric clinical psychologist were greatly beneficial to the child’s recovery. |
This patient is a 30-year-old male who presented to our emergency room following a head-on motor vehicle accident as a restrained driver. He described pain in his right ankle immediately after the accident and was taken to the emergency department as a trauma alert. ATLS protocol was instituted but no other injuries were identified. The patient’s right ankle was grossly deformed and locked in supination but skin was intact (Fig. ). The right lower extremity was neurovascularly intact. Initial X-rays (Fig. ) of the right ankle demonstrated a medial subtalar dislocation without associated fractures. In the emergency department, closed reduction was attempted under conscious sedation but was unsuccessful. Therefore, risks, benefits and alternatives were discussed and the patient elected to proceed with urgent closed vs open reduction of the right subtalar joint and any other indicated procedures.\nOnce general anesthesia was induced, closed reduction was attempted using axial traction with the knee in flexion. Closed reduction was unsuccessful once again (Fig. ) so the decision was made to attempt open reduction. An anteromedial approach was used dissect down to the talonavicular joint. Retractors were placed with care to avoid injury to tibialis anterior tendon or superficial peroneal nerve. The joint capsule was seen to be disrupted and further exposure revealed a thin, coronal fracture of the anteromedial aspect of the talar head measuring roughly 2 cm in length and 6 mm in thickness (Fig. ). Once this fragment was removed, the subtalar and talonavicular joints were easily reduced. It was thought that this incarcerated fragment was the likely cause for the unsuccessful closed reduction. The talar head fragment was unamendable for stabilization due to its small size with poor healing potential, thus it was removed. The talonavicular joint was then percutaneously pinned in retrograde fashion with three K-wires (Fig. ) to maintain anatomic reduction of the subtalar and talonavicular joints and the foot was splinted. At 2 weeks follow-up the patient was doing well and radiographs revealed no interval loss of reduction (Fig. ). |
A 64-year-old man reported to the Department of Prosthodontics, Faculty of Dental Sciences, Sri Ramachandra University, Chennai, India, with the chief complaint of an unstable loose mandibular denture. The medical history of the patient revealed that he was diabetic, hypertensive, and under medication. The patient presented with a history of neuromuscular incoordination for the past four years. He also complained of difficulty moving his jaws, normally being a complete denture wearer for the past seven years, leading to difficulty in chewing and speech, primarily due to loose lower dentures. His past medical history was found relevant for this case report. Manipulation with removable dentures, particularly with complete ones, is based on a very complex pattern of neuromuscular coordination. The denture, in itself a lifeless and passive implement, is completely useless unless operated by the neuromusculature. Diabetes mellitus is known to produce tenderness of the mucosa, rendering it prone to infections and dryness of the oral mucosa and glossodynia. The patients will complain of a burning sensation beneath the dentures, which they usually attribute to the dentures and futilely try to relieve the symptoms by making new ones. On clinical examination, the maxillary residual alveolar ridge was rounded and well formed, but the mandibular residual ridge was unfavorable due to a high degree of resorption (classified as Atwood’s Order V - low and well-rounded) [] (Figure ).\nThe treatment approach for this patient was to construct a mandibular denture using the conventional neutral zone technique and to use improvised procedures to minimize the chairside visits for the patient.\nThe objectives of the treatment are rehabilitation with complete denture therapy in a patient with poor neuromuscular coordination using an improvised neutral zone technique to achieve maximum prosthesis stability, comfort, and function; locating the neutral zone and arranging the denture teeth accordingly; and minimizing the ongoing diminution of the residual alveolar ridges. Figure reveals an orthopantograph of a severely resorbed mandibular arch.\nThe orofacial musculature plays a substantial role in developing the external polished surface of the denture and teeth arrangement. Forces developed during various muscular functions like chewing, speaking, and swallowing vary in direction and magnitude in each individual and in dissonant periods of time []. This is particularly notable in patients with neuromuscular incoordination, which is recorded by the neutral zone impression.\nClinical Visit 1\nDuring the patient’s first visit, as described in the conventional neutral zone technique, a preliminary impression of the maxillary and mandibular edentulous residual ridges was made with irreversible hydrocolloid impression material (Zhermack Dust-free Thixotropic Tropicalgin, Zhermack SpA, Badia Polesine [RO], Italy). The impressions were immediately cast in dental plaster (Bombay Burmah Trading Corporation, Ltd., Mumbai, India), and primary casts were prepared. Custom trays were fabricated with DPI (Dental Products of India) - RR cold cure acrylic material (Bombay Burmah Trading Corporation, Ltd., Mumbai, India). On the same day, border molding was done with admix material – three parts by weight of impression compound and seven parts by weight of tracing compound (DPI - Pinnacle Impression Compound and Tracing Sticks, The Bombay Burmah Trading Corporation, Ltd., Mumbai, India) [].The secondary impression was also made with the admix material (Figure ).\nThe master casts were poured in dental stone (Zhermack Elite Model Stone, Zhermack SpA, Badia Polesine [RO], Italy) and record bases were constructed with self-cure acrylic resin for the maxillary cast and heat-cure acrylic resin for the mandibular cast to improve record base stability. The wax occlusal rim was fabricated for the maxillary arch. A record base over the mandibular cast was fabricated with self-cure acrylic pillars.\nClinical Visit 2\nThe maxillary occlusal rim was inserted and parallelism was verified using the Fox occlusal plane. The mandibular record base was also placed in the patient’s mouth and checked for extension and stability by guiding the patient to perform mandibular movements. Once the mandibular record base was stabilized, the vertical jaw relation was determined with the help of self-cure acrylic resin vertical stops (3 mm × 8 mm) placed on either side of the mandibular canine-premolar region.\nThe patient was made to sit in an upright position and two prominent points were marked on the patient’s face - one on the nose and one on the chin. The vertical dimension at rest (VDR) was checked between these two points with the help of a divider and a 12-inch ruler. The determined VDR was 7.1 mm. Vertical dimension at occlusion (VDO) was determined with the help of self-cure acrylic stops fabricated on the mandibular record base. The patient was instructed to bite on the acrylic stops as it reached the early dough stage along with the maxillary occlusal rim, which was visualized and checked with the help of the divider and 12-inch ruler (Figure ).\nThe established VDO was 6.8 mm. Once the resin was set, the excess resin was trimmed off, thus culminating the vertical jaw relation.\nThe horizontal jaw relation was recorded using the admix material. This admix material is manipulated in the patient’s mouth at around 40° C. The patient was instructed to perform routine mandibular movements (including swallowing, sucking of the lips, and pronouncing the vowels), which aided in molding the neutral zone space (Figure ).\nThe external surface was completely contoured by the orofacial musculature.The maxillary and the mandibular rims were fused at the centric relation.\nThe maxillary and mandibular occlusal rims were articulated in a mean value articulator to fabricate indices surrounding the neutral zone plaster impression on the mandibular cast ( Figure ).\nThree notches were made on the cast: one in the anterior and two in the posterior regions. This was followed by applying separating medium on the cast, the record base, and over the neutral zone record. Boxing was done with modeling wax, and plaster of Paris was poured into the boxing up to the upper surface. The plaster indices were sectioned into a labial and buccal index and a lingual index in order to guide the removal and placement of these indices. The neutral zone record is then removed, and the acrylic stops are trimmed off from the denture base. Separating medium was applied on the inner surfaces of the indices which were then reassembled. Wax was poured in the space representing the neutral zone, forming the new occlusal rim on the mandibular record base. Figure shows the occlusal rim created and the plaster index.\nThe mandibular teeth were arranged following the index, and the maxillary teeth were arranged following the mandibular teeth arrangement. In order to preserve the contours established by the plaster indices in the neutral zone, no additional wax added to the denture flanges.\nClinical Visit 3\nA wax try-in was performed to evaluate mandibular record base stability, aesthetics, and intraoral occlusion. The patient successfully performed all the movements mentioned earlier. The trial dentures were processed with heat-cure acrylic resin. The denture was polished so that the customized contours remained unaltered.\nClinical Visit 4\nThe mandibular denture was again evaluated with the plaster index prior to denture insertion. (Figure )\nThe denture was inserted and verified for retention, stability, and occlusion. The patient was comfortable with the complete denture prosthesis. Periodic recall visits were scheduled to verify the retention, comfort, and function. |
A 77-year-old female with hypertension, untreated hyperlipidemia, hypothyroidism, but without prior history of CAD or angina symptoms was referred to a cardiologist's office for a treadmill exercise test secondary to new onset palpitations. She denied any chest pain or pressure, shortness of breath, exertional dyspnea, or leg swelling. She quit smoking 36 years ago and has no family history of early cardiovascular diseases. She has a very distant cardiac work-up years ago, including a stress test and an echocardiogram, which the patient reported were unremarkable. Vital signs prior to the test were a blood pressure of 140/78, heart rate of 80, and a respiratory rate of 14. Physical exam was unremarkable except for a systolic ejection murmur that was graded II/VI at the base. EKG was at baseline with a normal sinus rhythm, normal axis, and occasional premature ventricular complexes (PVCs).\nThe patient underwent an exercise stress test using the Bruce protocol and was able to complete stage 1 with exercise for three minutes at a speed of 1.7 mph and a 10% incline. The test was terminated due to dyspnea and fatigue without chest pain. She reached a heart rate of 141 beats per minute which was 98% of predicted for her age. She accomplished 4.5 metabolic equivalents of exertion. With exercise, she had occasional atrial premature complexes and PVCs with a ventricular couplet in recovery. She started to notice tightness in her chest. Her peak blood pressure at the time was 218/90.\nThe patient was transferred onto a stretcher, and an IV line was started. She was given sublingual nitroglycerin, 325 mg of aspirin to chew, and one 5 mg IV push of metoprolol tartrate. She then received nitroglycerin paste and metoprolol tartrate IV every 5 min for two more doses. At that time, her EKG on the stretcher showed ST elevations in leads I, aVL, V5, and V6 with ST depressions in leads III, aVF, and V1-V3 consistent with a lateral wall evolving myocardial infarction (). She was transferred urgently to our institution for cardiac catheterization.\nThe patient underwent an emergent cardiac catheterization with left ventriculography and intravascular ultrasound (IVUS) within 2 hours after onset of symptoms. Troponin-I levels prior to the catheterization increased to 11.17 (normal less than 0.05 ng/ml). The rest of the laboratories were within normal limits including a thyroid-stimulating hormone (TSH) level. Coronary angiography showed nonobstructive coronary artery disease (pLAD 40%) and highly tortuous coronary arteries. IVUS of the proximal LAD revealed a minimal lumen area of 5.2mm2, and no ruptured plaques. Left ventriculogram revealed a left ventricular ejection fraction (LVEF) of 20% and severe mid-cavitary hypokinesis with basal and apical hyperkinesis (Figures and ). To our knowledge, this is the first case of treadmill exercise testing-triggered mid-left ventricular ballooning variant of takotsubo cardiomyopathy, whereby obstructive epicardial CAD and ruptured plaques were excluded with angiography and IVUS, respectively.\nThe patient was started on medical management with standard therapy for heart failure. A follow-up echocardiogram was done two days after the event which redemonstrated mid-left ventricular ballooning, with an improved LVEF of 35%. The patient remained asymptomatic during the course of her hospitalization and troponin levels trended down from a postcardiac catheterization peak of 16.06 ng/ml. An echocardiogram was repeated during an outpatient follow-up two weeks later which showed resolution of wall motion abnormalities and an LVEF of 45-50%. |
A 28-year-old woman with a previous diagnosis of neurofibromatosis type I (NF I) presented to the hospital with dizziness, nausea, emesis, and anorexia. Magnetic resonance imaging of her head demonstrated a left thalamic mass and obstructive hydrocephalus. Seven years prior, the patient underwent ablation of atrioventricular nodal reentry tachycardia. Electrophysiology study at that time demonstrated dual AV nodal physiology, and she underwent successful cryoablation of her slow pathway without further history of palpitations. The ablation was complicated by biventricular failure requiring inotropic infusion for 12 hours. Rapid resolution occurred, and the patient was discharged without further event.\nThe patient underwent a biopsy of the left thalamic mass and drain placement to relieve obstruction. During anesthesia induction supraventricular tachycardia (SVT) occurred and was terminated with cardioversion. The procedure was aborted, and the patient was returned to the intensive care unit. A postoperative echocardiogram demonstrated severe left and right ventricular function and hypokinetic basal left ventricular segments, with akinesis of all other segments. The EF was less than 10% (see echocardiogram in Figures –). Inotropic support with milrinone was initiated. Three days later, limited echocardiogram demonstrated normal left ventricular size with hyperdynamic systolic function and an EF greater than 70% (see echocardiogram in Figures –). Metoprolol tartrate was initiated to prevent recurrence of SVT. The patient returned to the operating room and underwent successful external ventricular drain placement. Induction of anesthesia was uneventful. The patient was discharged without further cardiac event.\nThe patient again presented for symptoms with worsening hydrocephalus requiring further intervention. Upon anesthesia induction, the patient again developed SVT requiring cardioversion. Profound hypertension surrounded this arrhythmia with systolic blood pressure greater than 220 mmHg. Given the recurrent intraoperative hemodynamic events, testing of urinary and serum metanephrines was pursued which demonstrated elevated metanephrines and catecholamines. CT scan demonstrated a heterogeneous enhancing mass of the left adrenal gland and dotatate scan demonstrated increased uptake in the left adrenal gland suggestive of pheochromocytoma. Prazosin was initiated prior to definitive treatment of the hydrocephalus and pheochromocytoma. She underwent robotic-assisted laparoscopic left adrenalectomy in addition to left ventriculoperitoneal shunt placement. Pathology confirmed pheochromocytoma. The patient was discharged without further event and with normal left ventricular function. |
In August 2010, a 57-year-old Chinese male presented with epistaxis and decreased hearing for 1 month. No additional symptoms, such as a neck mass, nasal obstruction, headache, diplopia or other cranial nerve palsies, were noted. The patient had no history of previous or synchronous tumours or any family history of cancer. Nasopharyngoscopy revealed a large exophytic tumour that was covered by smooth mucosa, which grew from the right posterolateral nasopharyngeal wall in the right posterior naris. Magnetic resonance imaging (MRI) scans of the nasopharynx and neck using gadolinium enhancement demonstrated a 2.0 × 1.5 × 2.0 cm well enhanced mass over the right posterior nasopharynx with right retropharyngeal node enlargement. The tumour extended across the right parapharyngeal space and infiltrated into the medial pterygoid muscle. In addition, skull base erosion was detected with right alar lamina involvement (Fig. ). Cervical lymph node metastasis was not observed. Hematologic, hepatic and renal function tests revealed no abnormalities. The patients underwent chest and abdomen computed tomography (CT) as well as a bone scintigram, and no distant metastasis was found. A biopsy of the nasopharynx was performed.\nIn the biopsy specimen, normal salivary tissue was not present. The tumours were ill demarcated without encapsulation. Tumour cells were arranged in nests and nodules. Two morphologic patterns of the tumour cells were observed. Some small round cells exhibited dark nuclei and scant cytoplasm. Other large cells contained round to oval pale nuclei and eosinophilic to amphophilic cytoplasm. In the central region of the tumour cell nests, large cells displayed a solid growth pattern. Small dark cells were clustered at the periphery of the tumour cell nests and appeared palisaded. Prominent nucleoli and mitosis can be observed, and an average of three mitotic figures were observed per 10 high-power fields (original magnification × 400).\nIn the immunohistochemical analysis, the tumour cells were immunoreactive with P63, vimentin, and cytokeratin (CK7 and CK14) antibodies and focally immunoreactive with a calponin antibody. This case of BCAC was not positive for smooth muscle actin or CD117. The proliferative index as demonstrated by Ki-67 was approximately 10%. Based on the immunohistochemistry results and the pathological findings, which included tumour islands with solid proliferation, basaloid-like cells containing large pale and small dark cells, an infiltrative margin, cellular and nuclear pleomorphism, and prominent mitosis, the patient was diagnosed with a solid-type minor salivary gland BCAC (Fig. ).\nBased on the 2002 American Joint Committee on Cancer (AJCC) Tumor, Node, Metastasis (TNM) staging system [], the tumour was classified as stage III (T3N0M0).\nIn our case, the patient received intensity-modulated radiation therapy (IMRT) with 6 MV X-rays. The delineation of the gross tumour volume (GTV) was based on the primary tumour volume determined from the physical and imaging examinations. The clinical target volume (CTV) was defined as the whole nasopharyngeal cavity, the clivus, the skull base, the pterygoid plates, the parapharyngeal space, the sphenoid sinus, the posterior one-third of the nasal cavity, the maxillary sinus, and the drainage of the upper neck (levels II, III, and Va. A total dose of 70.4 Gy/32 F/6.2 W was administered based on the planning target volume (PTVg) (GTV with 0.5 cm margin). The PTV60 was defined as 60 Gy/30 F (CTV with 0.5 cm margin) (Fig. ). After radiotherapy, MRI and nasopharyngoscopy revealed complete disappearance of the tumour (Fig. ). The patient was followed up every 3 months for the first 2 years, every 6 months for another 3 years, and then every 12 months. A follow-up at 72 months did not detect any evidence of disease recurrence. The patient developed moderate mucositis as an acute adverse event. However, he did not exhibit any grade 3/4 late adverse events, such as xerostomia, dysgeusia, or hearing impairment. |
An 86-year-old woman presented with acute severe back pain with no neurologic deficit. Plain X-ray, computed tomography (CT) scan, and magnetic resonance imaging (MRI) () showed acute extension-distraction fracture of the T12 body with the intravertebral cleft and segmental lordotic change of the thoracolumbar spine. There were also multiple planar vertebral bodies as a result of previous osteoporotic thoracolumbar fractures. The condition of the thoracolumbar spine was highly unstable, and the aggravation of extension-distraction of the fractured vertebral body was likely to lead to neurological deficits. We decided to perform surgical treatment with instrumentation and fusion for the long segments above and below the fractured T12. However, it was predicted that stable fixation of pedicle screws would not be easy and that postoperative pseudoarthrosis and instability were likely to recur, considering the deformed vertebral bodies around the fractured T12 and the bone mineral densitometry (BMD) findings (−6.7, lumbar spine). Most importantly, the patient was judged as inoperable due to her weak physical condition caused by greatly reduced cardiopulmonary function (originating from heart failure and restrictive lung disease), in addition to a weak constitution (a height of 140 cm and weight of 38 kg). After deep consideration, the patient and her family refused surgical treatment and decided that she should be treated conservatively. As conservative treatment, bedrest in the lateral decubitus posture and pain control were performed for the initial three weeks along with the use of teriparatide (subcutaneous injection, 20 μg/day) with calcium and vitamin D supplementation. Afterwards, sitting and standing with wearable orthoses were carried out gradually depending on back pain. On the follow-up radiological examinations (), the collapse of fractured vertebral body proceeds with reducing intravertebral cleft at 3- and 6-month follow-up CT scans. The calcification and fusion of the interspinous space were identified earlier than the formation of bone bridges around and inside the fractured vertebra. Bone fusion with bone bridging and ankyloses around the fractured vertebral body, correction of the initial segmental lordotic change, and disappearance of the intravertebral gap were noted at 1-year follow-up CT scan. She completely recovered from her back pain, and is carrying out self-walking and normal daily activities. |
A 22-years-old Caucasian female was referred to our department, complaining of a 5-year history of intermittent pain in her right knee. She did not suffer from any medical condition, she was not on any medications and never smoked. Her symptoms started insidiously, following a minor soft tissue injury (no fracture). On clinical examination, she was found to have a right knee effusion and limitation of movement (ROM 10-90°). She had severe pain over her knee. She could weight bear with significant discomfort. Her blood tests were all normal, including the coagulation screen and all inflammatory markers. The X-rays performed were unremarkable (). An magnetic resonance imaging (MRI) scan was performed which suggested the presence of a suprapatellar fluid collection with cysts arising from the synovium. Following these results, an arthroscopy was performed. The cysts were excised and biopsied. The biopsies revealed normal synovium. Following the procedure, the patient remained asymptomatic for approximately 3 years. She then returned with similar symptomatology. A second MRI was performed showed the presence of a suprapatellar fluid collection measuring 4.7 × 2.1 cm at the suprapatellar area. No hemosiderin was noted within the collection and no local erosion. Surgical exploration was performed, and a poorly encapsulated vascular mass was encountered extending from the distal belly of the vastus intermedius to the knee. Excessive use of electrocautery and ligation of the numerous vessels were performed during resection. The histological examination of the mass revealed a dilated vascular space containing fibrous papillary projections and organized thrombus. It was suggestive of IPEH (Masson’s Tumor) ( and ). The patient’s symptoms recurred 2 years later. An MRI scan was repeated showing a 1 × 3 × 10 cm cystic lesion of the distal anterior thigh. The patient was then referred to the vascular department for embolization of the vascular supply of the tumor. Following this procedure, the patient has recovered fully, and she now remains asymptomatic to date (5½ years follow-up). |
A 7-month-old ex-30-week preterm female infant presented to the emergency department of our hospital with a two-month history of worsening intermittent vomiting and failure to thrive despite nutritional optimization and trial of different infant formulas. Past medical history was notable for prolonged NICU stay mainly due to delays in oral feeding. She had no pulmonary, cardiac, or intestinal complications of prematurity. She did not require any surgical procedure or hospital admission following NICU discharge at around 6 weeks of age. History is also negative for recent fevers or recurrent infections.\nOn presentation, she was in no acute distress and had normal vital signs. Weight and height were both below the first percentile. Physical examination was significant for the presence of hepatosplenomegaly. Laboratory workup was notable for elevated transaminases and significant hypercalcemia (4.47 mmol/L; Normal Range: 2.12-2.74mmol/L). Baseline phosphorus level was normal and the parathyroid hormone level was appropriately suppressed as seen in . She was admitted to hospital for management and further workup.\nAggressive intravascular fluid resuscitation with normal saline only partially improved serum calcium levels. Furosemide and calcitonin were used in succession, but they also failed to have a noticeable impact on serum calcium levels. Two doses of pamidronate, 0.5 mg/kg each two days apart, were eventually successful in restoring normal calcium levels. She also switched to low calcium infant formula, Calcilo- XD.\nAn extensive workup for viral and fungal etiologies was negative, as was the evaluation for metabolic, genetic, and oncologic causes of hypercalcemia. The skeletal survey did not show any lytic lesions; CT scan of the chest, abdomen, and pelvis was negative for the presence of lymphadenopathy or pulmonary lesions. Hepatosplenomegaly was confirmed with a CT scan; however, the underlying pathology was not revealed until a liver biopsy was performed for persistently elevated liver enzymes and massive hepatomegaly. Liver biopsy showed lobular histiocytic infiltrate with well-formed granulomas, hemophagocytosis, and increased portal/periportal and pericellular fibrosis with bridging indicating chronicity, with no further evidence to indicate an underlying etiology of the granulomas.\nShe was discharged home after 3 weeks of hospitalization once adequate weight gain and normal and stable calcium levels were achieved. Liver enzymes were improved but remained elevated. She had required readmission after one month for the recurrence of vomiting and poor weight. She had no fevers. Workup on this admission showed leukopenia and return of hypercalcemia (). Chronic granulomatous disease, immunodeficiency, tuberculosis, and hemophagocytic lymphohistiocytosis (HLH) were considered in the differential; however, screening was negative. HLH genotyping showed only a single allele mutation on UNC13 gene, but this variant did not explain her findings. Despite the lack of a standard definition of infantile sarcoidosis, this disease was considered due to elevated levels of angiotensin-converting enzyme (ACE) at 108 U/L (18-90). In the light of negative results for a possible underlying immunodeficiency and malignancy, she was placed on prednisolone 1 mg/kg/day for hypercalcemia. She remained afebrile and was discharged home in stable condition with prednisolone.\nAfter 5 weeks of prednisolone treatment, she presented with daily emesis and low-grade fevers. Complete blood count showed pancytopenia, peripheral smear showed fungal elements. She was admitted to the intensive care unit for disseminated fungal infection. Urine Histoplasma antigen was found to be positive. Systemic antifungal treatment was started. Of note, serum calcium and liver enzyme levels were normal during the third admission (). Because of disseminated fungal infection, prednisolone was discontinued and a hydrocortisone taper was initiated. She had an excellent response to antifungal treatment. Hepatosplenomegaly resolved and all other serum markers have improved. She remained on systemic antifungal treatment for 9 months. She made full-recovery and caught up with growth and development. |
A 39-year-old white female patient was admitted to the emergency department of our hospital with symptoms of acute intestinal obstruction, first noted 48 hours before (colicky abdominal pain, vomiting, and abdominal distention). The patient was tetraplegic due to Friedreich's disease diagnosed 25 years ago and was hospitalized permanently in a center for chronic disorders.\nUpon physical examination, a prominent abdominal mass distorting her trunk shape was noted. In palpation, the abdomen had considerable distension with diffuse tenderness and moderate rebound tenderness. Despite the distension in the upper abdomen, a huge tumor could easily be palpated occupying mainly the lower abdomen.\nThe neurological examination revealed good responsiveness of the patient, quadriplegia, loss of tendon reflexes, and positive Babinski sign bilaterally. She was then referred for cardiac evaluation, where mitral insufficiency and left ventricular hypertrophy were found.\nThe white blood cell count was 18,100/μL with 79.1% neutrophils, which was further elevated the second day; urea was found 51 mg/dL and γGT 54 mU/mL. The rest of the blood tests were normal.\nThe most possible diagnosis was intestinal obstruction probably due to the tumor. An abdominal computed tomography was performed, which revealed a huge intra-abdominal mass originating most possibly from the right cornu of the uterus (Figures , , ).\nAn emergency exploratory laparotomy followed with a midline incision.\nAfter the peritoneal cavity was opened an enormous circumscribed mass measuring 29.5 × 22 × 12 cm was revealed displacing the bowel to the abdominal periphery. The mass originated from the right cornu of the uterus. After lysis of the adhesions between the tumor and the greater omentum, the tumor was excised () and the right uterine cornu was ligated with a nonabsorbable suture. Inspection of the small intestine revealed a stenotic area in its central portion due to an adhesion with the omentum. The adhesion was taken down and careful hemostasis followed. Finally the abdominal wound was closed in the ordinary fashion.\nThe tumor, as well as the peritoneal washings, was sent for histological and cytological assessment, which did not reveal any evidence of malignancy. Histological assessment of the specimen revealed a uterine leiomyoma with increased mitotic activity. The postoperative course was uneventful, and the patient had no further complications. At present, two years after surgery the patient fares well and lives in a center for chronic disorders. |
This study was approved by the Ethics Committee of Orthopedic Surgery Department, Imam Khomeini Hospital, Tehran, Iran and a written consent was signed by the parents.\nA 7-yr-old boy, the only child of otherwise healthy parents was referred the Pediatric Orthopedic Clinic, Imam Khomeini Hospital, Tehran, Iran on July 2018 with the diagnosis of CP. The reason for referral was the parents’ concern about the increasing severity of disease despite regular occupational therapy.\nOn physical examination, the patient was developmentally delayed, unable to walk or stand, with obvious cognitional and gross and fine motor retardation. Flexion contractures were noted in elbows, wrists, knees, and hips. There was bilateral equinovarus deformity of feet and increased popliteal angle. Plantar reflexes showed extension response and DTRs were exaggerated. Spastic response of muscles was recorded after continuous stretching. Sitting balance was extremely unstable ().\nThe patient was the result of a consanguine marriage and normal pregnancy. Birth weight was 2950 gr and head circumference and height were 35 and 47, respectively. The few first months of his life showed normal weight gaining and development. He was able to hold his head in 5 months and roll over at 7 months age. The first time the parents had been told about the possibility of an abnormality was in a routine screening at 5 months age. The pediatrician noticed a decreased head circumference growth. Further investigation showed the head circumference reached a plateau (40 cm) in its growth around 12 months age (). His general and developmental condition seemed to experience a sudden pause with progressive delay in growth and development since then. He lost his ability to rolling over and never gained any gross motor milestones. His face became expressionless and his eyes started to sink into the orbits (). Other findings were: apparent cachectic dwarfism, microcephaly, loss of facial adipose tissue, pigmented retinopathy, thoracolumbar kyphosis, multiple joint contractures, senile appearance, photosensitivity, and thin and dry hair.\nAlthough physical examination had a lot of similarity to a patient with CP, the history was inconsistent with the diagnosis of CP in its almost all aspects. This made us reevaluate the diagnosis. After a thorough history taking, some clues were added to our knowledge which was critical to the correct diagnosis. These include rapid regression of all motor functions, regression of language and fine motor functions and facial changes which are not compatible with CP.\nAt 7-yr-old age, he was in a cachectic dwarfism condition. The progeroid appearance narrowed our differential diagnosis.\nOur first diagnosis based on clinical findings and progression of the disease was Cockayne syndrome. The diagnosis was later confirmed by molecular analysis for Cockayne syndrome. The patient was homozygous for ECCR6 gene (genotype: c.2551 T>A /p.W851R- c.2551 T>A /p.W851R). The parents were also heterozygous for the same gene. This was also true for the patient’s only sister. |
A 40-year-old woman with stage IIb right invasive ductal carcinoma underwent bilateral skin-sparing mastectomy and immediate subpectoral tissue expander breast reconstruction in anticipation of right breast adjuvant radiation (). Nine months following completion of radiation therapy, she elected to undergo bilateral DIEP flap breast reconstruction. She had a body mass index of 25 and no other significant medical history or contraindications to surgery. Computed tomography-angiography of the abdomen showed acceptable perforators for reconstruction.\nHer DIEP reconstruction was uneventful. Single-perforator flaps were raised in both hemiabdomens on large periumbilical perforators. The abdominal flaps were transferred to the contralateral chest with a 90° rotation. The left hemiabdominal flap perforator was located 4.8 cm to the left of the umbilicus and 2.3 cm caudal. The right hemiabdominal flap perforator was located 5.3 cm to the right of the umbilicus and 0.6 cm caudal. Clinical and indocyanine green laser angiographic examinations of the fully dissected abdominal flaps showed excellent perfusion. On each side, a single venous anastomosis was performed with 3.0-mm coupler between the larger medial vena comitans and the anterograde internal mammary vein at the level of the third rib. Flaps were placed in the prepectoral plane. Lower pole was heavily radiated, and poorly expanded mastectomy skin was excised. An identical procedure was performed on the contralateral nonradiated breast for symmetry.\nCutaneous Doppler flap checks proceeded postoperatively, with noted strong arterial and venous signals. On postoperative day 3, the patient got out of the shower and was noted to have acute venous congestion of the right flap. A venous signal at this point was still noted and the congestion resolved by the time the operative team was assembled 1 hour later. Upon ambulation postoperative day 4, she had recurrent venous congestion and loss of venous signal and was urgently taken to the operating room for exploration ().\nExploration of the flap proceeded as follows: Upon releasing her superior flap incision, the congestion resolved and a venous signal returned. The vascular anastomosis was inspected for thrombosis, kinking, twisting, tension, compression from hematoma, all of which were excluded. Continued monitoring of the flap intraoperatively did reveal that the venous signal was lost when the superior mastectomy skin was reapproximated to the flap skin paddle.\nObservation revealed that the perforator may have been kinking between the pectoralis muscle and the remnant abdominal fascial cuff around the perforator. Despite making a full-thickness myotomy in the pectoralis muscle under the perforator, the venous signal was again lost when the mastectomy skin was opposed to the flap. The flap inset was therefore left open, and the cause of congestion was determined to be compression ().\nThe flap completely recovered and had a normal examination until 5 weeks postoperatively when the patient presented again with acute venous congestion (). Her superior mastectomy skin had been closed in clinic 2 days prior. The congestion was noted to resolve in the supine position and again upon release of the superior incision. The patient was then monitored in the hospital with leech therapy for 5 days. The flap congestion completely resolved, and the patient had no further complications or fat necrosis of the flap. Her open wound was allowed to heal by secondary intention, and she was offered scar revision but declined. Her final result can be seen in over 1 year later. |
An 82-year-old male was admitted to the emergency department for worsening shortness of breath and hypoxia. He was admitted a week after he was diagnosed with a left ninth rib fracture secondary to a fall. He had long-standing history of chronic obstructive pulmonary disease, coronary artery disease, and peripheral vascular disease. Chest radiographs revealed a left pleural effusion and possible infiltrate. The patient was initially treated with a nebulizer, prednisone, and empiric antibiotic coverage with ceftriaxone and azithromycin. The patient failed to improve with the medical interventions and a therapeutic thoracentesis was performed. The thoracentesis was completed with ultrasound guidance, with the puncture made above the 11th rib at mid chest on the left. The pleural effusion was found to be frank blood. No immediate complications were noted, and the patient was taken to recovery. The next day the patient was found to be in respiratory distress. A chest x-ray revealed an opaque left hemithorax that was likely rapid accumulation of pleural fluid (Fig. ). A follow-up contrast-enhanced computed tomography (CT) of the chest performed during the arterial phase revealed a left intercostal pseudoaneurysm with hemothorax and adjacent compressive atelectasis (Fig. ). Ultrasound of the left chest wall was performed (Fig. ) directly over the thoracentesis site and doppler flow revealed bidirectional fluid flow, indicating the presence of a large pseudoaneurysm (Fig. ).\nFollowing identification of the left intercostal pseudoaneurysm, the patient underwent a thoracic aortogram and multiple-level left intercostal angiogram (Fig. ) under IV conscious sedation. Selective catheterization of the T5, T6, and T7 intercostal arteries was unsuccessful in identifying the pseudoaneurysm. Selective catheterization of T10 and T11 intercostal arteries was performed with a C2 Cobra catheter, following multiple catheter exchanges due to the patient’s atherosclerotic vessels. The pseudoaneurysm was ultimately found to have a left T10 origin and the C2 Cobra catheter was exchanged for a microcatheter. Once access was gained, coil embolization of the pseudoaneurysm was performed with a series of 15 Axium micro coils. Significant room was left on both sides of the pseudoaneurysm and a follow-up angiogram was performed via the microcatheter, then a 5-French Cobra catheter. The follow-up angiogram demonstrated no further filling of the pseudoaneurysm (Fig. ). The catheter was removed and a Perclose device was placed in the left groin for hemostasis. Following completion of the procedure, the patient was taken to recovery. The patient proceeded to return to his baseline following medical management during the remainder of his hospital stay and was discharged home after 5 days. |
A 51-year-old female with a history of rheumatoid arthritis and a 10.5-pack-year smoking history presented with an aspergilloma in her right lung. After failing medical management, she was treated with a right pneumonectomy at an outside institution. This was complicated by BPF and empyema of the pneumonectomy cavity. She underwent two additional thoracotomies requiring rib resection, and placement of serratus anterior and later latissimus dorsi flap to close the fistula. Seven months following her last operation, she presented to us with stridor, persistent cough, and dysphagia, concerning for postpneumonectomy syndrome. Review of last computed tomography (CT) imaging from three months after the pneumonectomy revealed a multiloculated pleural space, with air fluid levels in the pneumonectomy cavity. An updated CT scan showed interval progressive rightward mediastinal shift with nearly complete obliteration of the pneumonectomy cavity by the heart (Fig. ). A bronchoscopy was performed, which demonstrated narrowing of the left mainstem bronchus (Fig. a) and stenosis of the lower lobe bronchial orifice due to external compression of the airways. Results of a previous complex right-sided BPF with two areas of disrupted bronchial staple line were noted (Fig. b).\nThe patient elected to proceed with operative correction of her postpneumonectomy syndrome. A thoracotomy in the fifth intercostal space was performed and dense adhesions in the chest with rotation of mediastinal structures were faced. Upon entering the pleural space, a small loculated serous fluid collection was encountered. To rule out an infected field, the pleural rind and fluid samples were sent for intraoperative gram stains, which returned negative. Cultures were also submitted. The mediastinum was mobilized from the chest wall, taking care to avoid damage to the muscle flaps, which had previously sealed the BPF. As a result, only the mid and inferior portion of the mediastinum was mobilized. A saline immersion test was performed to ensure the integrity of the muscle flap seal over the right mainstem bronchus. The implants were sized based on measuring the amount of saline instilled in the chest, and close hemodynamic monitoring of arterial and central venous pressures. Before placing the implants, the thoracotomy was closed temporarily after placement of implant sizers, monitoring hemodynamics to ensure there was no right heart compression. Two implants (250 mL and 100 mL) were placed into the pleural cavity, and the thoracotomy was closed. The postoperative recovery was uneventful. The patient was discharged on post-operative day 5. She noted complete resolution of her stridor, cough, as well as dysphagia four weeks post-operatively. Her post-operative chest radiograph showed partial medialization of the inferior mediastinum with persistent rightward deviation of the proximal trachea (Fig. ). At time preparation of this manuscript, the patient continues to have full resolution of symptoms at fourteen months following surgery. |
Female patient, aged 30 years, reported dissatisfaction with the protrusion of teeth and lips. The frontal facial analysis showed symmetry, balanced facial thirds, good proportion between facial height and width, characteristics of mesofacial individuals. The lateral facial analysis revealed a Pattern I face, with good convexity, well-positioned maxilla and mandible. Lateral evaluation of the lower facial third evidenced increased projection of the lips, which compromised the facial esthetics. Evaluation of smile revealed that the upper arch presented good exposure of the upper incisors and gingiva, with excess exposure of lower incisors and asymmetry of the lower lip. It also revealed a good vertical relationship between the upper incisors and upper lip ().\nAnalysis of dental arches showed Angle Class I malocclusion, with excellent molar, premolar and canine sagittal relationships; however, with absence of the upper right first molar. Non-coincident upper and lower dental midlines were observed, due to deviation of the upper midline to the left because of a greater crowding of tooth #22 and contra-angulation of tooth #21. The overjet and overbite were reduced, due to the increased inclination of upper and lower incisors (). The upper arch presented moderate crowding and rotation of teeth #11, #21 and #22. The lower arch presented good alignment and leveling, with presence of a fixed canine-to-canine retainer on the lingual aspect, due to a previous orthodontic treatment ().\nAnalysis of the panoramic radiograph showed absence of the upper right first molar and upper and lower third molars. The other teeth and periodontal structures were in normal condition ().\nThe initial cephalometric analysis revealed good positioning of the maxilla and mandible, slightly divergent angles of the palatal, occlusal and mandibular planes, and normal lower facial height, characteristic of mesofacial individuals ().\n» The upper incisors had a slightly increased inclination (1.PP=115°). This angle assesses the relationship between the long axis of incisors and the palatal plane, with a mean normal value of 110°. » The upper incisors had a good vertical relationship with the upper lip, with a FAOP (Functional Aesthetic Occlusal Plane) of 2.5 mm. The lower incisors required extrusion of 2.5 mm to touch the FAOP plane (FAOP=+2.5 mm/-2.5 mm). The FAOP evaluates the positioning relationship between molars, incisors and upper lip stoma. The normal measure is 2.0 to 4.0 mm with the upper incisor. The lower incisor must be tangent to this plane. » The lower incisors had a slightly increased inclination (IMPA=100°). This angle assesses the relationship between long axis of lower incisors and the mandibular plane, and the normal measure is 90°. » Retromolar space is the space between the distal aspect of the crown of the lower second molar and the mesial aspect of the mandibular ramus. This space must be compatible with the need for distalization (). There was good space in the retromolar region (RMR) to perform distalization of the lower arch.\nThe orthodontic treatment planning consisted of alignment and leveling the arches and subsequent retraction, with anchorage on intra-alveolar orthodontic miniscrews, which were positioned between the first molars and second premolars, with the objective of reducing the protrusion and inclination of incisors and consequently improving facial and smile esthetics.\nInteractive self-ligating brackets, 0.022-in slot, with MBT prescription were used. This prescription was selected due to the greater torque in upper incisors (central incisors + 17° and lateral incisors + 10°) to obtain greater torque control during retraction, since the upper incisors needed small reduction in their inclination. In the lower arch, torques are reduced (lower incisors -6°), with less torque control, to allow greater reduction of inclination during retraction.\nThe alignment of the upper and lower arches was initiated with 0.014-in thermoactivated NiTi archwires, followed by 0.018-in; 0.014 x 0.025-in and 0.018 x 0.025-in ().\nThe retraction of both arches began with the 0.019 x 0.025-in stainless steel archwires, anchored in orthodontic miniscrews positioned between the second premolars and first molars, on the buccal side. The ideal initial force for total retraction of the arch is 250g/cm² and it should gradually be increased in the following consultations, up to a maximum of 400g\n,\n. This calibration was performed by reducing the spring length. On the upper right side, a milder force was used due to absence of the first molar, offering less resistance to distalization in this hemiarch. The miniscrews used (SIN, Sistema de Implante Nacional S.A., São Paulo/SP) had 1.6-mm diameter, 8.0-mm body length and 1.0-mm transmucosal profile. They were placed at 8.0 mm towards the apex in relation to the main archwire in the upper arch, and at 7.0 mm in the lower arch. This positioning was performed according to the band of keratinized mucosa, which is narrower in the mandible than in the maxilla. The miniscrews were inserted in the mucogingival line (which separates the keratinized from the alveolar mucosa). They were placed with an inclination of 80 to 90° in relation to the cortical plate in the maxilla and more inclined in the mandible ().\nAfter six months of retraction, a mild reduction in the inclination of incisor crowns was clinically observed, with consequent reduction in inclination of the lips. This allowed greater interaction by the patient during treatment, who could assess the gradual alteration of the profile and give an opinion on the best time to complete the arches retraction - unlike with premolar extractions, in which total space closure is necessary and often requires changing the anchorage strategy for mesialization of posterior teeth, when the retraction of anterior teeth is no longer desired.\nRetraction of the lower arch was performed faster than that of the upper arch, generating an increased overjet. At that moment, the screws in the mandible were removed and the retraction was continued in the upper arch ().\nAfter three months of upper retraction, the overjet was normalized, and the canine relationships finalized with an key of occlusion. At that moment, the implant was placed in the region of the upper first molar. After the osseointegration period, the crown was placed on the implant ().\nSimultaneous retraction of the arches anchored on orthodontic miniscrews was able to retract the upper incisors in 2.3 mm and reduce their inclination by 5° (1.PP=110°). The lower incisors retracted 3.0 mm and reduced their inclination by 10° (IMPA=90°). The upper incisors, despite the retraction and reduction of inclination, maintained their relationship with the FAOP at 2.5 mm. This fact was due to retraction with intrusion vector. The lower incisors were also retracted, with a reduction in inclination; however, they extruded and touched the FAOP (FAOP = +2.5 mm/0.0 mm). Canines, premolars and molars ended in an key of occlusion. The incisor relationship improved, increasing the overjet and overbite ().\nIn the facial aspect, there were small positive changes, with a slight reduction in lip projection, compatible with the small reduction in incisor inclination ().\nAnalysis of the final panoramic radiograph did not show any significant alteration in relation to the initial radiograph, except for implant placement in the region of the upper right first molar ().\nCephalometrically, the most relevant changes were reduction of bimaxillary protrusion and inclination of the upper and lower incisors; distalization of all posterior teeth; maintenance of vertical dimension; and improvement of soft tissue esthetics. There was a 16° reduction in the interincisal angle, changing from 111° to 127° (). |
A 52-year-old man was admitted to the emergency room (ER) with a chief complaint of swelling in the right submandibular area, which was noticed a week earlier. Clinical examination revealed swelling in the right submandibular area and subglossal area. Computed tomography (CT) in the ER revealed abscesses in the right submandibular and subglossal areas (). At this time, the patient did not have any symptoms of breathing difficulty and did not show signs of upper airway obstruction on plain neck radiograph and neck CT imaging (). The patient was diagnosed the right submandibular abscess and right subglossal abscess, for which intraoral incision and drainage was performed after intravenous administration of antibiotics. The patient and his guardian were explained the possibility of airway obstruction, if symptoms deteriorated in the future, and the patient was transferred to the general ward for follow up observations for infection. The day after admission, the patient complained of mild swallowing and breathing difficulties, and his oxygen saturation level (SpO2) was 97%. Nasal cannular was used to administer 3 L/min of oxygen, and the process was monitored. However, the patient continued to experience breathing difficulties and the severity increased. After 2 hours, his vital signs were as follows: blood pressure of NIBP_SBP was 164 mmHg and NIBP_DBP 98 mmHg; pulse rate was 130 beats/min; respiratory rate was 28 breaths/min; body temperature 38. 4℃; and (SpO2) level was 97%.\nThe patient was consulted for airway evaluation. Partial airway obstruction was observed on endoscopic airway examination performed at the ENT department, and because the patient complained of severe breathing difficulties when lying flat in the supine position, he was instructed to maintain Semi-Fowler's position. For airway management, after discussion and consultation with the Departments of ENT and Anesthesiology, a decision was taken to secure the airway via intubation. The patient and his guardian were explained the tracheostomy procedure, possibility of an additional incision and drainage, and the patient care in the intensive care unit (ICU). After the obtaining the necessary consent, the patient was moved to the operation room (OR). During the pre-anesthetic evaluation in the OR, the anesthesiologist was of the opinion that the swelling in the oropharynx and hypopharynx regions were severe and stated that endotracheal intubation via the oral or nasal cavity would be difficult. Consequently, the ENT department was requested to perform a tracheostomy without preceding endotracheal intubation, which was to be performed under local anesthesia with oxygen being administered while the patient was in Semi-Fowler's position. In the OR, with the patient in Semi-Fowler's position and SpO2 level maintained at 99%, an ENT surgeon performed the tracheostomy. After 20 minutes from the initiation of tracheostomy, the patient suddenly showed difficulty in breathing and respiratory arrest, while his SpO2 level began to rapidly decrease, and even reaching 0%. That event took place during the lateral dissection of soft tissue including of muscles. The time taken from initiation of breathing difficulty to intubation after tracheal opening was 10 minutes after 30 minutes (min) from the incision of tracheostomy. The SpO2 level increased again once oxygen was supplied by placing a tracheal tube, but the maximum level reached was 80%, with no further increase. A right-sided pneumothorax was identified on the chest AP radiograph taken to assess the cause insufficient oxygen saturation following tracheostomy and intubation status (), whereas the findings on the preoperative chest radiograph were normal. The emergency thoracentesis was performed 30 minutes after identifying right-sided pneumothorax with the chest tube in the right lung region. A maxillofacial surgeon performed incision and drainage on the submandibular space through a neck and intraoral approach, after which the patient was moved to the ICU. An additional chest AP radiograph taken in the ICU, which showed a pneumothorax in the left lung region as well as right lung (). Thoracentesis was performed on the left lung region additionally ().\nThe patient was then in a state of stupor, and electroencephalography results indicated a mild to moderate diffused cerebral dysfunction, based on which hypoxic brain damage was diagnosed. The patient continued to show improvement and there, he underwent rehabilitative therapy with a diagnosis of quadriplegia from hypoxic brain damage. In the fifth month of hospitalization, the patient improved and was discharged. At the time of discharge, the patient showed a pattern of neural recovery, and it was recommended to the patient and his family that he actively needs to continue rehabilitation therapy to strengthen the muscles in his limbs and for his speech therapy. |
In March 2011, a 60-year-old woman presented at the gastroenterology unit of our hospital (AZ Nikolaas, Sint-Niklaas) with gastrointestinal bleeding (table ). Her medical history reported a deep venous thrombosis with lung embolism in 1999, gallbladder lithiasis, uterus myomatosus and a psychiatric disorder (anxiety and depression). Gastroscopy revealed esophageal varices that were successfully ligated. In addition, a CT scan of the abdomen showed a large tumor (diameter approximately 10 cm) in the left hypochondriac region, located cranial to the tail of the pancreas and expanding into the spleen area (fig. ). The tumor contained multiple calcifications in the spleen area and around the fundus of the stomach. A pancreatic tail tumor extending into the spleen or a primary splenic tumor was suspected. There was no invasion of the kidneys or adrenal glands, no retroperitoneal adenopathy, no parenchymal metastasis in the liver and no bone lesions. A diagnostic laparoscopy was performed. Anatomopathological examination of limited biopsy material showed the presence of a tumor with morphological and immunohistochemical features suggesting metastasis or extension of a clear cell renal cell carcinoma in the pancreas, although the possibility of an adrenocortical carcinoma could not be excluded. Hematoxylin and eosin staining revealed a lesion with a solid and nested growth pattern containing cells with clear cytoplasm, well-defined cell borders and small, slightly pleomorphic nuclei (fig. ). The tumor showed a strong positive reaction to staining for vimentin (fig. ) and focal positivity for CD10 (fig. ). It also showed strong staining with pancytokeratin (CK AE1/AE3) and focal positivity for synaptophysin; there was no immunoreactivity for CD45 (LCA).\nDue to the widespread nature of the tumor in the upper abdomen and the inconclusive anatomopathological examination, the patient underwent surgery in a university center (UZ Leuven). In June 2011, the tumor was resected with splenectomy and distal pancreatectomy. The anatomopathological examination was compatible with a NET originating from the pancreatic tail, which could have arisen from an intrasplenic pancreatic remnant. Microscopically, the tumor was strongly vascularized, with a heterogeneous population of tumor cells and varying atypical nuclei. Immunohistochemical analysis showed diffuse membrane expression of prekeratin and inhibin, partial expression of epithelial membrane antigen, a focal, dot-like expression of CK7, a weak partial expression of synaptophysin and a strong diffuse expression of CK20. A focal expression of MelanA was also observed, while neurofilament and calretinin were not expressed. The tumor had invaded the spleen and peripancreatic fat, which was resected in toto. The tumor was diagnosed as a stage II pancreatic NET, with a Ki67 index of 10%. Since the PET/CT scan in the initial staging showed no metastases, no treatment was initiated but a close clinical, biochemical and radiological follow-up was planned.\nIn October 2011, an increase in the level of the tumor marker chromogranin A was noted during follow-up laboratory testing. A CT scan of the abdomen revealed a hypercaptating lesion of approximately 3.5 cm, located at the residual part of the pancreatic corpus, without enlargement of the pancreas. Due to its hypervascular character, a residual tumor or tumor relapse was suspected. Because of this early relapse after extensive surgical resection, we initiated treatment with a LAR formulation of octreotide (Sandostatin LAR® 30 mg, Novartis), administered intramuscularly once a month. Extensive analysis of CT scan images after 3 months of treatment was planned.\nThe control CT scan in January 2012 revealed multiple small lesions with early arterial enhancement in the parenchyma of the liver, indicative of hypervascular metastases (fig. ). However, the patient presented no symptoms and biochemical tests showed normal liver function. Due to difficulties with the administration of the Sandostatin LAR® injections (the nurses reported difficulties with handling the syringes and with making a homogeneous solution), the treatment was switched to lanreotide Autogel® 120 mg (Somatuline® Autogel®, Ipsen NV), administered once a month. This long-acting somatostatin analog is provided as a prefilled syringe and is administered deep-subcutaneously in the upper outer quadrant of the buttocks. No difficulties with the administration of lanreotide Autogel® 120 mg were encountered, and the patient reported no side effects. Follow-up visits with CT scans were performed every 3 months. The disease at the residual pancreas and liver remained stable until November 2013. Chromogranin and neuron-specific enolase levels were also assessed and were found to be normalizing.\nDeterioration in the clinical status of the patient and weight loss were noted from November 2013 on. A CT scan revealed an increase in the size of the liver lesions and presence of peritoneal metastases with a low level of ascites (fig. ). A chemotherapy course consisting of 6 cycles of cisplatinum-etoposide was initiated in December 2013. This treatment was chosen above newer targeted therapies, such as sunitinib and everolimus, because of the aggressive evolution of the pancreatic NET in our patient. The chemotherapy was administered every 3 weeks according to the following schedule: day 1, cisplatinum 80 mg/m2; day 1, 2 and 3, etoposide 100 mg/m2. The lanreotide Autogel® 120 mg injections were continued during the chemotherapy course. After 3 cycles of chemotherapy, we saw a rapid decline in the quality of life of our patient and she refused any further treatment; the lanreotide injections were also stopped. Palliative care at home was initiated. The patient died due to clinical progressive disease 1 month after the chemotherapy had been stopped. |
A 26-year-old male, a native of Rajasthan, presented to the dermatology OPD with complaints of swelling over his upper lip of 6 month's duration. The patient had initially noticed a small red raised lesion on the middle part of the upper lip which was not painful, not itchy, and not fluid-filled. The swelling progressively increased in size over a few weeks and then spontaneously ruptured in the center, forming an ulcer. There was crusting and bleeding from the ulcer. There was no history of fever, loss of weight, cough, or other respiratory complaints. There was no history of travel to any country endemic for leishmaniasis. Treatment with multiple drugs, including antibiotics, by various medical practitioners had not caused regression of the lesion.\nOn examination, the vital parameters were normal. There was no hepatosplenomegaly. All other systems were also normal. Local examination of the face revealed a 4 × 3 cm nodular, indurated, swelling with well-defined edges over the upper lip and extending into the labial mucosa []. The center of the nodule was ulcerated and tender, with overlying crusting and pus discharge. There was a solitary 1 × 1 cm enlarged, nontender, submental lymph node on the left side. A clinical diagnosis of cutaneous leishmaniasis was made, with insect bite reaction and cutaneous tuberculosis kept in mind as remote possibilities.\nInvestigations revealed a total leukocyte count of 7800/mm3and absolute eosinophilic count of 1050/mm.[] All other hematological, biochemical, and radiological investigations were normal. VDRL and ELISA for HIV were negative. Slit-skin smear from the lesion showed lymphocytes with plasma cells, but no diagnostic clue was obtained. Skin biopsy from the edge of the lesion showed well-defined lymphocyte granulomas, with few plasma cells []; however no definite diagnosis could be made. As we were not able to confirm the diagnosis with these usual tests, we carried out fine needle aspiration cytology (FNAC) from the draining lymph node. Slides from the aspirate showed macrophages filled with Leishman-Donovan (LD) bodies []. A few LD bodies were seen outside the cells also. The diagnosis of cutaneous leishmaniasis was thus confirmed.\nWe treated the patient with intralesional sodium stibogluconate once a week for 4 weeks and tablet ketoconazole 400 mg once daily for the same duration. Two weeks after the start of treatment, the lesion had dried up and had started regressing in size []. The lesion completely regressed, without any residual scarring or pigmentation, over 4 weeks. The patient has now been followed up for 1 year and shows no signs of relapse. |
A 72-year-old Caucasian female presented to the emergency department with two days of nausea and vomiting and on examination a large palpable left iliac fossa mass. The patient however described normal bowel actions over the preceding two days and was pain-free.\nHer past medical history included a recent total knee arthroplasty (TKA) four weeks previously and the commencement of rivaroxaban, a direct Factor Xa inhibitor, a new anticoagulant which had been used to treat deep vein thrombosis (DVT) which had occurred postoperatively on the knee replacement side. Other comorbidities included hypertension, hypercholesterolaemia, and osteoarthritis which she was on medication for.\nPrior to surgery, a noncontrast computed tomography (CT) scan of the abdomen and pelvis showed a large amount of free fluid, around the liver and descending colon, within the pouch of Douglas, and bilaterally in the lower pelvis, including within a large left indirect inguinal hernia sac. shows a snapshot of the CT scan performed showing the large fluid-filled indirect inguinal hernia sac. The presence of a fluid-filled level within the hernia sac and free fluid within abdomen created concerns of a strangulated hernia, and the patient was scheduled for an urgent open left inguinal hernioplasty.\nHer preoperative status was within normal physiological parameters (blood pressure 132/70, heart rate 82 beats/minute, and oxygen saturation 100% in room air), hemodynamically stable with a hemoglobin level of 10.6 g/dL prior to surgery. Rivaroxaban was ceased prior to surgery.\nAt surgery, a left inguinal incision was performed, with exploration of the inguinal canal. A large indirect hernia sac was identified with a firm consistency with its contents. Opening of the sac revealed a large volume of blood and clot. The bowel was otherwise viable with no sign of perforation or other intra-abdominal contamination. A left inguinal hernioplasty was performed with a standard Lichtenstein repair.\nShe spent the subsequent two days as an inpatient under observation. She did not require blood transfusions during this period. Her hemoglobin levels over these two days fell to 93 g/dL and subsequently to 83 g/dL the day after. She was subsequently discharged home without further event.\nPostoperative review at two weeks did not show any abnormalities and subsequent intravenous contrast CT scan of her abdomen at 3 weeks was performed to ensure no obvious intraperitoneal cause for the bleeding. There was no evidence of residual hematoma, or any evidence of intra-abdominal lesion which may have accounted for unexplained intra-abdominal bleeding. |
A 40-year-old male patient was hospitalized for sharp, lancinating pain in his left loin, which was disrupting his daily routine for last 2 days. A week ago, the patient developed vesicular rash in the same area and was treated with topical and oral acyclovir for 1 week. The patient also experienced a burning sensation at the site of rash, which responded to etoricoxib 120 mg daily given for 5 days. The patient reported back after 5 days to the out-patient clinic due to persistent and severe left loin pain and was hospitalized. The investigations revealed normal hematology and biochemistry profile. Chest X-ray and ultrasonography (USG) abdomen were also normal. Serological tests for hepatitis B virus, human immunodeficiency virus, Epstein-Barr virus and herpes simplex virus were normal. herpes zoster virus IgM titer was 1:40 by immune fluorescent assay (normal value < 1:10). The patient was diagnosed as a case of post herpetic neuralgia and received intramuscular tramadol 50 mg as and when required. Etoricoxib was stopped and pregabalin 75 mg twice a day and paracetamol 1 g 3 times a day was started orally. In view of inadequate pain relief, the dose of pregabalin was increased to 150 mg twice a day. Remarkable improvement was noticed after increased dose of pregabalin. Hence, patient decided to go home. He was advised to continue pregabalin 150 mg twice a day and paracetamol orally whenever necessary.\nAfter 2 weeks, the patient came with complaints of low grade fever, itchy skin rash and swelling of lips and face since 3 day. Purpuric rash was observed on the face and exfoliative lesions were seen on the hands and feet, without any joint or mucosal involvement. He had raised erythrocyte sedimentation rate, C-reactive protein, peripheral eosinophilia (absolute eosinophil count of 1400/cm) and normal leukocyte and platelet counts. Dengue antigen (NS1) and malaria rapid tests were negative. Liver function tests revealed alanine transferase (ALT) 250 IU/L and aspirate transaminases (AST) 322 IU/L with normal bilirubin, alkaline phosphatase and gamma glutamyl transpeptidase, Anti-nuclear antibodies were negative. Ultrasonography did not reveal any liver abnormality. Serum pregabalin was 5 μg/mL; (N < 10 μg/mL; MedTox Lab Inc. USA, liquid chromatography/tandem mass spectrometry [MS]). The diagnosis of probable hypersensitivity to pregabalin was made.\nThe drug was stopped and patient was treated with topical betamethasone cream and oral prednisolone 40 mg daily for 10 days tapered over a period of 1½ months. He recovered completely. The casualty assessment by World Health Organization Uppsala Monitoring Center scale[] and Naranjo's algorithm[] (score 6) showed a probable relationship between the drug and the reaction. |
A 41-year-old female was reviewed in the Neurosurgical Clinic upon referral from ENT colleagues. The patient was undergoing radiological surveillance for follicular thyroid cancer for which she had undergone a thyroidectomy 13 years ago. She underwent a limited scan which included her anterior skull base, and subsequently had a complete MRI scan with contrast, as described below. Of note in the patient’s medical history, she had the pathological involvement of her uterus, thyroid, and breast, all of which form parts of the major criteria for CS diagnosis [,] Interestingly, not only is the patient a mother to two daughters, both of whom have been genetically confirmed as CS-positive, but she was herself the daughter of a CS patient. Moreover, this awareness from our Genetic Services Division led to the initial investigations whereby the patient’s system-wide pathologies were brought to light.\nThe purpose of the patient’s present follow-up was to monitor the remission status of thyroid cancer; however, she was found to have three incidental meningiomata. The patient denied any headaches, seizures, or other problems that could be attributable to the meningiomata. She did not have any visual problems and was neurologically intact.\nAn MRI brain scan in October 2018 revealed a small left frontal contrast-enhancing dural based lesion consistent with a meningioma of just over 3 cm x 1.5 cm, a left temporal convexity tiny meningioma, and a small left-sided sphenoid wing meningioma (Figures , , ). The left frontal convexity meningioma was associated with a small amount of edema but there were no other concerning features. Her case was discussed at the NeuroOncology multidisciplinary team meeting, as is our practice for all newly diagnosed tumors. Owing to the multiplicity of lesions, the small individual volume size, and the small collective volume, she was considered a candidate for stereotactic radiosurgery. She remains well at one-year post Gamma Knife radiosurgery. |
A 9-year-old boy was referred to our pediatric department with symptoms of chronic fatigue, headache, intermittent nausea, and vomiting for 2 months. At admission a weight loss of 2 kg and intermittent subfebril temperature as well as an unspecific tremor of both hands was reported. He had a medical history of erythema migrans 2 years ago treated adequately with amoxicillin for 2 weeks. There was no history of trauma or stay in subtropical countries. Family history revealed a Waldenstrom’s macroglobulinemia of his father and paternal uncle. Clinically, only mild postural tremor of both hands and mild horizontal nystagmus with no other clinical symptoms was noticed.\nA cerebral magnetic resonance imaging (MRI) angiography was performed, showing 2 small acute cerebellar infarctions in the posterior inferior cerebellar artery area and occlusion of the basilar artery, highly suspicious of an underlying vasculitis ( and ). Conventional cerebral angiography detected a subtotal occlusion of the basilar and bilateral vertebral arteries with retrograde blood flow to the posterior cerebral circulation ().\nExtended immunological, hematological, and endocrinological laboratory evaluation revealed no abnormalities, among others normal blood count; negative C-reactive protein; normal erythrocyte sedimentation rate; normal renal, thyroid, and liver function; negative thrombophilic parameters; and negative immunological tests with no contribution for systemic lupus erythematosus, antiphospholipid syndrome, or other rheumatologic diseases (). Instead, Borrelia burgdorferi serology was positive for IgM and IgG, with confirmation of 4 positive bands in Western blot. A lumbar puncture was performed, showing lymphocytosis with increased protein and decreased glucose levels along with an intrathecal production of Borrelia burgdorferi IgM antibodies.\nTherefore, neuroborreliosis was assumed and treatment was started with intravenous ceftriaxone 80 mg/kg/day for 3 weeks in addition to oral acetylsalicylic acid (ASS) 5 mg/kg/day and prednisolone 1 mg/kg/day. Clinical symptoms subsequently improved and neurological examination returned to normal soon after therapy was started.\nA follow-up lumbar puncture 3 weeks later showed a reduction of pleocytosis and normalization of liquor glucose and protein. Two months later, MRI angiography showed the absence of cerebellar diffusion restrictions, but an unchanged presentation of the basilar and bilateral vertebral arteries occlusion. The prednisolone therapy was tapered over 3 weeks, whereas ASS therapy was continued. Three months later, a subsequent MRI angiography showed an unvaried occlusion of the basilar and bilateral vertebral arteries with no areas of new infarction (). Clinical and radiological follow-up 6 and 12 months after diagnosis showed a boy without any residual symptoms but an unchanged MRI angiography with ongoing vascular occlusion. |
During a 3-year period, a previously healthy 82-year-old male repeatedly consulted a general practitioner as well as a rheumatologist because of joint pains. The patient was eventually diagnosed with seronegative rheumatoid arthritis. During this 3-year period, the patient was treated with increased doses of PO prednisolone to 20 mg daily and, eventually, the addition of methotrexate gradually increased to 15 mg weekly. Two series of intra-articular injections of methylprednisolone were administered to several metacarpophalangeal joints of the right hand as well as the right shoulder.\nThe patient was later admitted to the emergency department at the University Hospital, Uppsala, Sweden, 1 week after the last injections, with complaints of fever, chills, myalgia as well as an open wound on the right hand but denied any respiratory symptoms. The wound had appeared approximately at the time of the latest methylprednisolone injections and was open and purulent, with swelling of the dorsal side of the right hand. The left elbow and the dorsal side of the left hand were also swollen. The patient was circulatory stabile and afebrile. Auscultation of the heart and lungs revealed no abnormalities. Plasma CRP-levels (301 mg/l) and total leucocytes (15 × 10(9)/l) were elevated.\nBlood and tissue samples were taken for routine cultures before treatment with cefotaxime 1 g IV q8h was started. Surgical exploration and debridement of the wound was performed upon arrival, revealing damage to extensor tendons of the forearm/hand. The swollen left elbow was also punctured and clear fluid was extracted and these particular symptoms were therefore suspected to be due to a non-infectious process, e.g. reactive arthritis. Several surgical revisions were performed during the first 10 days, due to the emergence of novel abscesses on the hands and forearms.\nAll cultures acquired prior to antibiotic therapy resulted negative. Due to non-satisfactory effect 11 days after admission, treatment was replaced with a combined therapy of imipenem + cilastatin (500 mg/500 mg IV q8h) and clindamycin (600 mg IV q8h). Supplementary cultures were taken, as well as biopsies subsequently analyzed for non-tuberculous mycobacteria (NTM) (culture, microscopy, and PCR), fungus (microscopy), Nocardia and Actinomyces (cultures). Samples were taken from wound material for bacterial 16S-RNA PCR. TBC-specific IGRA (QuantiFERON, Cellestis) turned out negative.\nAll further wound cultures as well as direct microscopy for acid-fast rods (mycobacteria ssp.) resulted negative. The 16S-RNA PCR did though indicated the presence of L. bozemanii in wound material. No reports of L. bozemanii causing soft tissue infections were previously reported and, therefore, NTM was still considered a possible cause and the therapy was now altered to target mycobacteria. A combination of moxifloxacin (400 mg PO q24h), amikacin (500 mg IV q24h), and clarithromycin (500 mg IV q12h) was introduced. Additional tissue samples were taken from the right upper arm and again 16S-RNA PCR was positive for L. bozemanii, and a Legionella-specific PCR was performed which also detected L. bozemanii DNA. Serological analysis was undertaken and compared to blood samples acquired on admission, which revealed a fivefold increase of anti-body titers of L. bozemanii and L. longbeachae serogroup 1 and 2 during a 12-day period. Treatment was now continued with moxifloxacin and clarithromycin.\nDue to inadequate improvement of the patient's general condition a chest X-ray was performed on day 16. This revealed widespread perihilar consolidations, interpreted as alveolar edema. During the next 2 weeks, repeated X-rays showed only minor improvement, and a CT scan was therefore performed, revealing bilateral alveolar consolidations as well as consolidations in the left lung. These abnormalities were interpreted by radiologists to be due to a previous infection. During this period, several novel abscesses appeared on both upper limbs, requiring additional surgical evacuation.\nHowever, the patient eventually did show clear general improvement as well as reduction of skin lesions and could be discharged from the hospital, after approximately 3 months. Treatment with moxifloxacin and clarithromycin was continued for a further 2 months. Follow-up examination was performed after a further 4 months. No new abscesses had developed and the previous lesions had completely healed. |
A 7-month-old male child, born normally at full term with no dysmorphic features, was admitted to the hospital because of a severe attack of bronchopneumonia. He had been suffering from severe upper airway obstruction since birth, resulting in poor oral feeding. He had a history of multiple hospitalizations. He had undergone surgery for bilateral inguinal hernia shortly after birth (postnatal day 9), with no postoperative complications. However, a few days after discharge, his parents had noticed recurrent noisy breathing, which increased with time. He was also suffering from apneic attacks with diaphoresis during feeding; hence, oral feeding was not possible. The child was readmitted to the hospital but due to the impossibility of oral feeding, he was discharged on nasogastric feeding (regular infant milk formula with medium-chain triglyceride oil as no ENDF was available in India at that time). After discharge, he had to be hospitalized repeatedly because of aspiration pneumonia. Laryngoplasty was performed but oral feeding was still not possible because of residual narrowing, and the child was once again discharged on nasogastric feeding with standard infant formula plus medium-chain triglyceride oil. However, no weight gain was observed up to 3 months of age.\nAt the time of the current hospitalization, the child was on nasogastric feeding. His weight was 5.130 kg, height 61 cm, head circumference 43 cm, and mid-upperarm circumference (MUAC) was 12.5 cm. No abnormalities were observed on chest radiography, chest computed tomography (CT), echocardiography, brain magnetic resonance imaging, barium meal, and milk scan. Blood pressure and serum electrolyte levels were also normal. However, bronchoscopy showed signs of laryngomalacia and CT of the paranasal sinuses revealed narrowing and near occlusion (II degree) of the oropharynx. Posteriorly placed adenoids were seen. Based on these observations and the fact that the child's weight was less than the 3rd percentile on standardized WHO growth charts, he was diagnosed with growth faltering due to severe laryngomalacia.\nThe patient was admitted to the pediatric intensive care unit (PICU) and kept there for 3 weeks. An ENT specialist was consulted, who advised that tracheostomy be performed provided the child showed good oropharyngeal coordination and satisfactory weight gain. However, the child was not able to gain weight. Moreover, feed volume could not be increased to facilitate weight gain as gastroesophageal reflux disease was suspected. Thus, it was decided to initiate nutritional management with ENDF. The aim was to achieve enough weight gain to perform tracheostomy without any adverse events as satisfactory weight gain can lead to spontaneous resolution of upper airway obstruction, as well as better immunity and normal neurodevelopmental outcomes. Initially, the child was administered a 60–70 mL feed of ENDF every 3 h, adding up to a total energy intake of 100 kcal/kg/day. Gradually, under the pediatrician's guidance and over a period of 1 week, the intake was increased to 160–170 kcal/kg/day. The feed was well-tolerated, with no signs of diarrhea, flatulence, or vomiting. The child gained 400 g over 2 weeks and weighed 5.530 kg at the end of the 3-week PICU stay. This was the maximum growth velocity (30 g/day) ever achieved in the child's life. As a result, tracheostomy was performed successfully.\nAt this point, the infant was shifted to the ward and kept there for 2 additional days before being discharged on nasogastric feeding with 160–170 kcal/kg/day of ENDF. At discharge, he weighed 5.445 kg. The drop in weight was because of the posterior rhinoplasty he had to undergo during the third week of hospitalization. After discharge, the child gained weight rapidly, reaching 6 kg at the 2-week follow up. He did not show any signs of intolerance to the formula and was thriving well. Finally, at 8 weeks after discharge, when the child was weighing 7 kg, the nasogastric tube was successfully removed. After the removal of the tube, the oral feeding was continued with ENDF and home-made foods. Alternate feeds were given between ENDF formula and home-made diet as recommended for age (7 months) wherein ENDF accounted for 360–480 kcals/day of the total caloric intake which composed of 3-4 feeds, the child gradually developed tolerance for semi solid home cooked foods. Even after 8 weeks post discharge, patient regularly visited for follow up and was on infant formula and home cooked meals till the age of 12 months, meeting his recommended daily allowance as designed for Indians. The child's growth chart is shown in . The child's parents were pretty satisfied and credited the ENDF formula based nutritional intervention, as the importance of weight gain was emphasized to them by many specialist during their past hospitalizations. |
A 34-year-old man with dizziness, dyspnea, and inability to swallow referred to the hospital emergency department. The patient presented with dizziness, followed by dyspnea, a gradual feeling of dryness in the throat, an impaired reflex, slow papillary reaction to light, a decreased eye movement, a decreased power of facial muscles and limbs, normal deep tendon reflexes (DTR), lack of focal neurologic signs, and respiratory failure. The patient reported the use of clay cheese in the week before referring to the emergency. In the laboratory evaluation, hematological and biochemical tests such as complete blood cell counts, and also serum level of sodium, potassium, blood sugar, BUN, and creatinine were normal. electrocardiogram, magnetic resonance imaging, and chest X-ray were also reported as normal. The patient was under supportive therapy, respiratory care, cardiac monitoring, and pulse oximetry after transferring to the intensive care unit (ICU).\nBased on the clinical suspicion to botulism, diagnostic evaluations, including the evaluation of serum samples, emission, and gastric juice, were done to confirm the BT. The patient’s history showed the use of cabbage salad, vegetables, and clay cheese in the last week. Thus samples of these foods were evaluated regarding the BT. The patient was treated with three doses of antitoxin. Neurology consultation was done a day after hospitalization. After three days, the patient was transferred to the infectious diseases ward. After 11 days and resolving of ptosis, the power of organs reached the normal state (5/5), and the gag reflex was decreased; consequently, he was discharged with medication orders. Four days later, the BT test in blood and cheese samples came positive (toxin B) while it was negative in other food samples.\nThe patients’ wife also referred to the hospital with asthma and a feeling of dryness in the throat, followed by the inability to swallow, feeling of vomiting, and trouble speaking for four days. During admission to the emergency, the patient was conscious without a fever but, with dysarthria and bilateral ptosis, an impaired gag reflex, slow right papillary reaction to light, a decreased eye movement, a decreased power of facial muscles and limbs, normal DTR, and lack of focal neurologic signs and respiratory failure. This patient underwent supportive therapy and antitoxin prescription. In addition to these two patients, seven other members of the family referred to the hospital with the same signs of botulism to be treated. |
In late 2016, a 61 year old non-smoking female presented with exertional shortness of breath, mild pedal edema, distended neck veins, and a recent weight gain of 15 lbs over the prior 2 weeks. It was initially suspected that the patient was experiencing pulmonary embolism based on her clinical presentation and elevated D-dimer, but further testing revealed that she was experiencing cardiac tamponade with mild pulmonary hypertension and heart failure. Pericardiocentesis was performed without definitive diagnosis. In February 2017, the patient exhibited worsening respiratory symptoms without fever, hemoptysis, sputum production, B symptoms, or extremity edema. Baseline chest PET-CT images were reviewed by radiologists at both UCLA Oncology and Texas Tech University Health Sciences Center, demonstrating a large middle mediastinal mass encasing the main pulmonary artery, with pericardial and left pleural effusion (Figure & Figure ). Scattered pulmonary nodules and hypodense lesions were identified in the right lobe of the liver, consistent with a diagnosis of metastatic disease (Figure ). Biopsy of the mediastinal mass revealed a high grade undifferentiated malignant neoplasm composed of highly proliferative (ki67 staining ~50%) pleomorphic anaplastic epithelioid malignant cells with large areas of necrosis and fibrosis. Immunohistochemistry revealed strong antigenicity for CD31 and CD34, and weak antigenicity for D2-40 and Factor VIII indicating a diagnosis of angiosarcoma that was corroborated at both UC San Diego Health and MD Anderson.\nConventional treatment options were recommended, however the patient declined these based on low reported survival rates, and instead, requested the non-selective beta blocker propranolol as a single agent therapy. In May 2017, 40 mg/kg propranolol was administered daily and PET-CT scans were performed at regular intervals to assess the response of the tumor to propranolol. Assessment of tumor response was based on 18F-fluorodeoxyglucose (FDG) tracer uptake and measurements/assessments of the primary tumor and distant metastases. After 12 months of propranolol as a single agent therapy, significant debulking and decreased size of the residual mediastinal mass was observed on PET-CT scans, with resolution of pericardial effusion (Figure & Figure ). Pulmonary nodules were stable to regressed, and the nodules in the right lobe of the liver had completely resolved (Figure ). There was no evidence of residual hyper-metabolic activity based on FDG measurements in the primary lesion or in metastatic sites in the chest, abdomen, or pelvis on PET-CT. |
A 31-year-old HIV-infected black African female presented with an 8-day history of painful swallowing, sore eyes, malaise and a worsening rash. She had been on antiretroviral (ARV) regimen of nevirapine (NVP) 400 mg daily, tenofovir 300 mg daily and lamivudine 300 mg daily for more than 4 years uneventfully. She was on no other medications and had not taken any other medication in the preceding 8 weeks. She had acquired HIV via heterosexual contact – the exact date of HIV infection was unknown. At the time of initiation of ARV therapy, her nadir CD4 cell count was 139 cells/mm\n3 and her HIV RNA viral load at the time was unknown. Her last CD4 counts, done 31, 21 and 11 months before developing her current symptoms were 373, 356 and 387 cells/mm\n3 respectively. The last HIV RNA viral load test, done 11 months prior to the onset of her symptoms showed an undetectable viral load.\nShe gave a history of forgetting to take her ARV medication for a day. The following day, 10 days before development of her symptoms she took 2 days equivalent of her ARVs in one day, in her own words “to make up for the missed dose”.\nOn examination, she was normotensive and had a temperature of 38.8°C. She had conjunctivitis and hemorrhagic cheilitis but no involvement of the genital mucosa. She had epidermal necrosis involving predominantly her trunk and face and to a lesser extent palms, soles and extremities, totaling 10% of her body surface area, 3% of which was stripping. Initial laboratory studies showed normal blood count, except for an elevated eosinophil count of 0.70 × 10\n9/L. The liver and renal function tests were normal. Her ARVs were stopped and her epidermal necrosis did not extend and eosinophil counts normalized. Over the next few days her skin condition improved and she was discharged 10 days later fully re-epithelialized with the exception of the lips, which were still eroded in areas. On review, 2 weeks after discharge, her skin had normalized except for residual hyperpigmentation. She was started on a single daily tablet of Odimune\n®, a fixed drug combination of tenofovir 300 mg, emtricitabine 200 mg and efavirenz 600 mg. On follow-up, 4 weeks later she was tolerating the new ARV regimen. |
A 67-year-old man was admitted to our hospital because of liver dysfunction during a screening examination. Enhanced abdominal computed tomography (CT) revealed a hypervascular mass of 35 mm in diameter in the descending portion of the duodenum (Fig. ), and the left three sections of the liver were occupied by multiple cystic tumors with contrast enhancement of the cystic wall, 13 cm in diameter (Fig. ). A duodenal tumor was identified on gastrointestinal endoscopy (Fig. ), and a biopsy revealed a NET. The serum levels of insulin, gastrin, and glucagon were within normal ranges. CT did not initially reveal evidence of pancreatic invasion between the tumor and the pancreas; however, irregularities of the duodenal wall and swelling of the lymph nodes around the pancreatic parenchyma were observed. Thus, the patient was diagnosed with non-functional duodenal NET with multiple liver metastases, T2N1M1 stage IV (UICC 8th). In addition, CT revealed the anatomical variation of the CHA, which branched from the SMA and ran fully through the head of the pancreatic parenchyma (Fig. , Additional file Figure S1). The CHA branches into the left hepatic artery (LHA), the middle hepatic artery (MHA), and the right hepatic artery (RHA) (Fig. a, b). Furthermore, a developed gastric arterial arcade, 4 mm in diameter, was found between the left gastric artery (LGA) and the right gastric artery (RGA). The RGA was branched from a distal portion at a distance of 10 mm from the root of the LHA (Fig. ). Incidentally, we did not observe stenosis of the celiac axis due to compression by the median arcuate ligament. We planned PD and left trisectionectomy with caudate lobectomy combined resection of the tp-CHA with the preservation of the gastric arterial arcade in order to maintain arterial flow of the remnant liver, preserving the route of the celiac artery to the right posterior hepatic artery (RPHA) via the gastric arterial arcade from the LGA to the RGA, LHA, and RHA. If the hepatic arterial flow could not be maintained by this route, the preservation of the tp-CHA by separating from pancreatic parenchyma or arterial reconstruction using radial artery graft between CHA and RHA was planned. Four weeks after percutaneous transhepatic portal embolization, surgery was carried out.\nAfter laparotomy, the gastric arterial arcade was exposed and encircled, and the LHA, RHA, and proper hepatic artery (PHA) were encircled (Fig. ). The LHA was divided at the distal side of the origin of the RGA. The MHA and the right anterior hepatic artery (RAHA) were also divided. The left portal branch and the right anterior portal branch were divided (Fig. ). The liver was transected, and the left hepatic duct and right anterior hepatic duct were divided. The left trisections and caudate lobe were anatomically resected. After clamping the PHA, the hepatic arterial signals of the RPHA via the gastric arterial arcade were confirmed by intraoperative Doppler ultrasonography (Fig. ). After trisectionectomy and caudate lobectomy, PD was performed. The pancreatic head was dissected from the SMA after the upper jejunum was divided. The pancreas was divided in front of the SMV. Finally, the specimen was only connected by the tp-CHA and the common hepatic duct (CHD) (Fig. ). The hepatic arterial signals of the RPHA was maintained after clamping the PHA. The PHA and the origin of CHA were divided, and the tp-CHA was taken out with the pancreatic head (Fig. ). The CHD was divided, and the specimen was removed (Fig. ). Reconstruction was performed via modified Child’s method. The operative time was 1072 min and the intraoperative blood loss was 3052 ml, and red blood cell transfusion was performed (1680 ml).\nPostoperatively, the patient developed pancreatic fistula (Clavien-Dindo IIIa) and biliary leak (Clavien-Dindo IIIa), and these complications were treated conservatively. There were no signs of hepatic ischemia. The patient was discharged on postoperative day 39. The pathological diagnosis was duodenal neuroendocrine tumor G2 with multiple liver metastases. The Ki-67 labeling index was < 20%, and staining for chromogranin A and synaptophysin were positive. There was no evidence of invasion of the pancreatic parenchyma; however, the duodenal tumor was confined to the MP layer, and one of the 25 examined lymph nodes was positive, and moderate lymphovascular invasion was observed. The final diagnosis was pMP, med, INFa, ly1, v2, pPM0, pDM0, and pEM0. The patient has shown no recurrence in the 22 months since the operation. Enhanced abdominal CT at 4 months after surgery revealed the blood flow of the RPHA via the gastric arcade (Fig. ).\nOver the years, several authors have described variations in the hepatic arterial anatomy; a CHA arising from the SMA—called the hepatomesenteric type—is a rare clinical entity. Yang et al. and Hiatt et al. reported that this condition was observed in only 31 of 1324 patients and 15 of 1000 patients, respectively [, ]. A CHA passing through the pancreatic head parenchyma, tp-CHA, is even rarer; Yang et al. [] reported that among 31 patients with the hepatomesenteric type, only 3 had this condition.\nWhen PD is scheduled in such patients with tp-CHA, it is important to maintain the arterial supply to the liver. Surgeons should preoperatively determine whether to preserve or perform combined resection of the tp-CHA. Tp-CHA preservation was selected in several previous reports [, , ]. This surgical procedure is technically feasible; however, there is a risk of a positive surgical margin or insufficient lymph node dissection and a tendency for increased intraoperative blood loss during the separation of the pancreatic parenchyma. If the tp-CHA is resected, reconstruction is usually necessary in order to maintain the hepatic arterial flow. Previous reports [, , ] have described successful arterial reconstruction after CHA resection during PD; however, such procedures are associated with an increased risk of thromboembolism, which can lead to a fatal outcome, especially in HPD []. In contrast, when collateral circulation develops, surgeons can perform combined resection of the tp-CHA, preserving the collateral circulation without arterial reconstruction. Several reports have recommended preoperative embolization of CHA in order to maintain the hepatic arterial flow through enlarged collateral arteries []. Although preoperative embolization can increase the liver arterial flow through collateral arteries, it is not routinely recommended because of the risk of complications, which includes the migration of embolic material [, ].\nA developed gastric arcade or pancreaticoduodenal arcade is frequently seen in patients with the stenosis of the CHA due to factors such as compression by the median arcuate ligament []. There are only a few cases in which the hepatomesenteric trunk and the tp-CHA and the association between the tp-CHA and the development of a gastric arterial arcade have not been reported. On the other hand, Miyamoto et al. reported the case of a patient with pancreatic head cancer with a CHA arising from the SMA who underwent radical PD combined with the resection of the CHA, in which the hepatic arterial flow was maintained via the gastric arterial arcade []. In this report, the patient did not have a developed gastric arterial arcade; however, the hepatic arterial flow via the gastric arterial arcade was sufficient and hepatic ischemia was not detected after the operation. Considering this case, even if the patients with tp-CHA do not have a developed gastric arterial arcade, surgeons may be able to preserve hepatic arterial flow via the gastric arterial arcade alone. If the hepatic arterial flow via the gastric arterial arcade alone is adequate after clamping the PHA, the combined resection of the tp-CHA can be considered, even if the gastric arcade is not developed before surgery. In cases in which the hepatic arterial flow is not adequate, the preservation of the tp-CHA or arterial reconstruction should be considered.\nWhen performing HPD, a PD-first procedure before hepatectomy is generally performed, as this approach is anatomically rational []. However, in the present case, performing hepatectomy after PD carried a risk of the arterial supply to the liver being reduced during hepatectomy. Had we chosen a PD-first procedure and the hepatic arterial flow not been maintained after CHA resection, it would have been necessary to perform arterial reconstruction before liver transection. This method is associated with a risk of injury to the reconstructed artery and thrombosis during liver transection. Given the above, we opted to perform hepatectomy before PD in our patient with a tp-CHA undergoing HPD.\nIn the procedure for separating the tp-CHA from the pancreatic parenchyma entirely, the surgeon should be concerned about the increasing rate of hemorrhage, surgery time, and the risk of injury to the tp-CHA. The surgical reconstruction of the hepatic artery when performing HPD is also associated with a high degree of risk. The association between tp-CHA and gastric arterial arcade was recognized on preoperative CT scans; the development of this collateral circulation may have the potential to prevent ischemia-related liver complications. From these points of view, the preoperative identification of the developed arcade of the gastric arteries helps in planning an appropriate operative procedure, and this procedure seems to be a viable and simple option. To our knowledge, this is the first report of PD combined with resection of a tp-CHA without preoperative embolization. Furthermore, this is also the first report of HPD for a patient with a tp-CHA. The preoperative identification of the developed arcade of the gastric arteries helps in planning the appropriate operative procedure when PD is scheduled for patients with a tp-CHA. |
This study was approved by the Ethics Committee of Orthopedic Surgery Department, Imam Khomeini Hospital, Tehran, Iran and a written consent was signed by the parents.\nA 7-yr-old boy, the only child of otherwise healthy parents was referred the Pediatric Orthopedic Clinic, Imam Khomeini Hospital, Tehran, Iran on July 2018 with the diagnosis of CP. The reason for referral was the parents’ concern about the increasing severity of disease despite regular occupational therapy.\nOn physical examination, the patient was developmentally delayed, unable to walk or stand, with obvious cognitional and gross and fine motor retardation. Flexion contractures were noted in elbows, wrists, knees, and hips. There was bilateral equinovarus deformity of feet and increased popliteal angle. Plantar reflexes showed extension response and DTRs were exaggerated. Spastic response of muscles was recorded after continuous stretching. Sitting balance was extremely unstable ().\nThe patient was the result of a consanguine marriage and normal pregnancy. Birth weight was 2950 gr and head circumference and height were 35 and 47, respectively. The few first months of his life showed normal weight gaining and development. He was able to hold his head in 5 months and roll over at 7 months age. The first time the parents had been told about the possibility of an abnormality was in a routine screening at 5 months age. The pediatrician noticed a decreased head circumference growth. Further investigation showed the head circumference reached a plateau (40 cm) in its growth around 12 months age (). His general and developmental condition seemed to experience a sudden pause with progressive delay in growth and development since then. He lost his ability to rolling over and never gained any gross motor milestones. His face became expressionless and his eyes started to sink into the orbits (). Other findings were: apparent cachectic dwarfism, microcephaly, loss of facial adipose tissue, pigmented retinopathy, thoracolumbar kyphosis, multiple joint contractures, senile appearance, photosensitivity, and thin and dry hair.\nAlthough physical examination had a lot of similarity to a patient with CP, the history was inconsistent with the diagnosis of CP in its almost all aspects. This made us reevaluate the diagnosis. After a thorough history taking, some clues were added to our knowledge which was critical to the correct diagnosis. These include rapid regression of all motor functions, regression of language and fine motor functions and facial changes which are not compatible with CP.\nAt 7-yr-old age, he was in a cachectic dwarfism condition. The progeroid appearance narrowed our differential diagnosis.\nOur first diagnosis based on clinical findings and progression of the disease was Cockayne syndrome. The diagnosis was later confirmed by molecular analysis for Cockayne syndrome. The patient was homozygous for ECCR6 gene (genotype: c.2551 T>A /p.W851R- c.2551 T>A /p.W851R). The parents were also heterozygous for the same gene. This was also true for the patient’s only sister. |
A 21-year-old man with no significant past medical history presented to the emergency department (ED) with concerns of diarrhea that had started one month prior, and had worsened one week before the presentation. Initially, he had experienced an episode of diarrhea every other day, and then three to five episodes of watery brown diarrhea on a daily basis. Two days before admission, he had passed large amounts of bright red blood per rectum with associated non-bloody emesis. Over the previous month, he had lost weight, although he was unable to quantify the weight loss. He had no past medical history, had not undergone any surgeries, and not taken any medication besides occasional loratadine for seasonal allergies. He denied any relevant family history and was an occasional alcohol drinker, non-smoker, and had no history of illicit drug use. He had visited the ED only once previously, for diarrhea following a course of antibiotics for a nonhealing mouth ulcer.\nUpon presentation, the patient was tachycardic and tachypneic. Notable vital signs were a heart rate of 132 beats per minute and blood pressure of 87/72 mmHg. Oxygen saturation and body temperature were within normal limits. His laboratory investigation revealed a hemoglobin of 7.6 g/dL, leukocytosis (white blood cell count of 19.6 x 109/L), platelets elevated at 882 x 109/L, and elevated alkaline phosphatase (ALP) at 430 U/L. His fecal immunochemical test was positive for blood. He was given a bolus of Ringer’s lactate solution and started on empiric antibiotics. He was subsequently admitted for the management of acute blood loss anemia secondary to GI bleed and was transfused a total of three units of packed red blood cells. CT of his abdomen revealed pancolitis with a mildly distended gallbladder. Flexible sigmoidoscopy was performed on his first day of admission, and pathology revealed CD. He received three days of intravenous methylprednisolone, which was subsequently switched to oral prednisone 60 mg daily. After the initiation of the steroid regimen, he reported no episodes of bloody diarrhea. His hemoglobin eventually stabilized to be between 9.9 and 10.6 g/dL. His hemoglobin and hematocrit levels were monitored closely throughout the admission period. Liver function studies revealed persistently elevated ALP, and his gamma-glutamyl transferase (GGT) level was also elevated. Magnetic resonance cholangiopancreatography revealed a long segment stricture of the common bile duct. Endoscopic retrograde cholangiopancreatography (ERCP) on day four revealed that one-third of his common bile duct was dilated; mucus and sludge were present, as well as severe diffuse strictures, which were concerning for PSC (Figure ). Biopsies were taken during this procedure for cytology, and the patient was informed of these findings and the pathology results from the previous flexible sigmoidoscopy (Figures , ). He was educated on the importance of close follow-up with an outpatient GI physician.\nBy the morning of his discharge, his hemoglobin had stabilized, and he was no longer experiencing bloody diarrhea, nausea/vomiting, or abdominal pain. The patient was instructed to follow up in one week to initiate ustekinumab therapy, and he was discharged with a long tapering dose of oral prednisone. |
A 59-year-old female patient presented to a plastic surgery clinic with a lump on her right medial thigh. She noticed the lump eight months ago and it was gradually increasing in size. It was painless initially; however, the lump gradually became tender. The patient was otherwise fit and well with no co-existing morbidities. Closer examination of the right thigh revealed a high consistency mass, with reduced mobility. There was no right inguinal lymphadenopathy or lymph nodes enlargement.\nLaboratory blood tests were unremarkable. An MRI scan was requested to further assess the mass, which was irregular but well-defined in the medial compartment of the thigh, measuring 8.2 x 6.6 x 4.3 cm in size. The mass showed an intermediate signal on both T1 and T2-weighted imaging, appearing hyperintense compared to the adjacent muscular tissue, which also persisted on fat-suppressed imaging. The mass invaded the intramuscular fat planes and caused compression and displacement of the adductor muscles. There was no evidence of intra-muscular invasion. Normal cortical outline and medullary signal intensity of the femur were seen in the right thigh. There was no evidence of bone contusion, marrow oedema, fracture line, or cortical discontinuity. Other muscles of the thigh had a normal outline and signal intensity with no evidence of focal or diffuse oedema. Neurovascular structures were unremarkable. Overall, MRI findings were suggestive of soft tissue neoplasm, with a possibility of a neurogenic tumour. A wide excisional biopsy was then performed to further assess the tissue histologically by the pathologist and confirm the diagnosis. The removed mass measuring 8.5 x 6.5 x 5.4 cm and is shown in Figure . The mass had a smooth outer surface and a soft consistency. The cut surface showed a homogenous tan-white appearance with some slit-like spaces and occasional haemorrhagic spots. Multiple sections were processed for histopathological examination from different planes.\nHistopathology of the mass revealed a tumour arranged in sheets and fascicles composed of round to oval cells along with variably sized adipocytes (Figure ). Numerous interspersed lipoblasts with indented nuclei were also seen. Moreover, severe nuclear atypia was noted including bizarre cells (Figure and Figure ). An area of necrosis was seen with a few areas, which showed malignant and fibrous histiocytoma-like features. Furthermore, a few thin-walled dilated and congested blood vessels were also noted. Also, brisk mitosis was noted (35-38 per 10 HPF) (Figure ). Immunohistochemical staining (IHC) was also performed and the tumour cells were diffusely positive for vimentin and focally positive for S-100; while negative for creatine kinase (CK), smooth muscle antigen (SMA), desmin, CD34 and MyoD1. Overall, the pathological findings are all suggestive features of a high-grade sarcoma, favouring the epithelioid variant of PLS.\nThe patient was also referred to the Department of Nuclear Medicine & Molecular Imaging for post-surgical restaging and assessment for possible metastases using 18F-fluorodeoxyglucose positron emission tomography-computed tomography (18F-FDG PET-CT) scan. The abdominopelvic section showed the liver measuring within normal limits; however, the scan revealed an ill-defined hypermetabolic hypodense lesion seen involving segment VI measuring approximately 2.8 x 1.8 cm suggestive of hepatic metastases with concurrent fatty liver. The scan was also remarkable for the musculoskeletal system, revealing multiple hypermetabolic osseous lesions involving the skull base, right iliac bone, and few vertebrae (predominantly T7-L1); which was suggestive of skeletal metastases. A non-hypermetabolic pulmonary nodule in the right lung field was also detected; it was considered to be likely benign, but interval scanning is suggested to monitor further progress. Finally, there was also evidence of diffuse subtle hypermetabolism at the site of surgery which is suggestive of postsurgical inflammatory sequelae; however, microscopic residual disease cannot be completely ruled out. These multi-organ lesions were due to be biopsied to examine their immunohistochemical findings to confirm metastatic lesions of primary pleiomorphic liposarcoma. However, the patient did not attend the follow-up appointments. |
Diana (a pseudonym) is a young woman of 25 years old that reached the Eating Disorders Centre, Division of Endocrine and Metabolic Diseases, San Luca Hospital in Milan, following a dramatic weight loss. Diana reached the Centre with a BMI of 16.06 kg/m2 reporting several disruptions in her eating patterns and several distressful alterations in her body image perception. As reported in the clinical history, Diana’s first eating related crisis was dated back 2 years before her current admission, with a subtle episode when she started a diet to lose some weight after health issues related to her thyroid. During that period, Diana was located abroad for work and – under moderate stress – she began a restrictive diet with a low caloric intake that brought her to lose 10 kg in 6 months. Diana’s weight remained constant in the following months but she developed an obsessive attention to the caloric intake along with intrusive thoughts regarding her weight and regarding specific types of foods. Diana also reported body-related image distortions such as overvaluation of her weight, mirror and body checking, and avoidance of body exposure. Moreover, during the crises, she reported frequent crying spells observing her body in front of a mirror.\nIn the last year, Diana reported a stressful situation at the University that heightened her psychological symptoms. Following these new difficulties, Diana re-enacted the restrictive conducts, reducing the caloric intake with a consequent weight loss of 4 kg in a month. In the period before the admission, the restrictive conduits were accompanied by self-induced vomit and daily binge episodes.\nDiana matched all the DSM-5 criteria for a diagnosis of AN, binge-purge subtype. Compatible with the diagnosis, Diana presented distortions in her body perception and obsessive thoughts regarding her weight and her body image; for these reasons, she was considered as an optimal candidate for the interoceptive assessment.\nAt the begin of the rehabilitative protocol, Diana’s blood panels showed no signs of metabolic distress, with values in normal ranges. At her admission, Diana’s thyroid levels were within normal range and they remained within the normal range during the curse of the treatment. Endocrinologist suggested a chronic autoimmune normal-functioning condition. The psychiatric assessment indicated mood alterations toward a depressive condition accompanied by severe sleep difficulties and insomnia.\nThe rehabilitative program was composed of a multidisciplinary approach that included several experts in different fields: endocrinology, psychiatry, psychology, and nutrition. The specialists collaborated in an outpatients service tailored to the specific users’ needs. The rehabilitative program could extend from two to four cycles of treatment. Diana followed a two-cycle rehabilitative protocol with a frequency of 3 sessions a week for a total of 37 sessions. The protocol was composed of psychological intervention with group psychotherapy and individual sessions focused upon a psyco-corporal therapy approach (body-oriented psychotherapy). Psychological intervention was accompanied by psychiatric and pharmacological support (citalopram and mirtazapine), to moderate Diana’s mood alterations. The rehabilitative protocol was integrated with alimentary education sessions provided by the nutritionist. Additionally, the protocol was also accompanied by a nutritional program with fixed meals (both in quantity and composition) that Diana consumed under supervision. Scheduled assessment sessions ensured an adequate monitoring of the progress.\nFrom the beginning, Diana showed a deep insight regarding her condition. Nonetheless, from her first session, Diana showed severe difficulties in following the assigned rehabilitative diet due to obsessive thoughts connected to her body weight and to certain types of food. In the following weeks, Diana improved her adherence to the recovery protocol with a better ability to follow the changes in the diet both on quantitative both on the qualitative level (e.g., types of foods consumed). Diana reached a BMI of 19.00 kg/m2 at the end of her second cycle of rehabilitative treatment. Considering the noticeable improvements regarding her eating behaviors and her general clinical condition, Diana was dismissed from the Centre and continued her program following only monthly assessments accompanied by individual psychotherapy. |
The first patient was a 60-year-old male who presented at our institution with complaints of exertional dyspnoea and angina. He was a known case of interstitial lung disease and was dependant on continuous oxygen support (5 L/min) for more than 20 h a day, to maintain a saturation of 90%. He was assessed for lung transplant. Cardiac catheterisation study revealed high pulmonary artery (PA) pressures. The pulmonary vascular resistance (PVR) was high (3.7 wood units). The coronary angiogram showed insignificant coronary artery disease. Dobutamine stress echocardiography confirmed good left ventricular function without any ischaemia. A High-resolution computed tomography scan of the lungs revealed bilateral interlobular interstitial thickening with honeycombing pattern of bronchiectasis and mediastinal lymphadenopathy. Lung perfusion scan revealed relatively reduced perfusion in the left lung contributing to 30% of the total pulmonary function. Pulmonary function tests revealed a significant restrictive pathology with a forced expiratory volume in 1 s of 1.26 L (27% of predicted). He underwent right single-lung transplantation under cardiopulmonary bypass. In view of high PA pressures, cardiopulmonary bypass was initiated after heparinisation with an arterial cannula in aorta and direct right atrial cannulation for venous access. He had a total bypass time of 198 min and total ischaemic time of the graft was 180 min. He was shifted to Intensive Care Unit (ICU) with stable haemodynamics and minimal ionotropic support. He was extubated on the same day of surgery and had an uneventful post-operative period. The post-operative bronchoscopy showed healthy bronchial stump. The patient was provided with regular respiratory care and was shifted to the ward on the 3rd post-operative day. His endotracheal secretions showed heavy growth of pseudomonas and the growth was adequately managed with injection piperacillin–tazobactam. He recovered well with a smooth post-operative course and was discharged on the 17th post-operative day with an improved saturation of 98%, requiring minimal support of O2. |
A 27-year-old Emirati male presented to Cleveland Clinic Abu Dhabi (CCAD) emergency department (ED) for the first time in Sept 2015 complaining of severe abdominal pain. The pain has been episodic for the last 4 years and had significantly affected his work and family life. He was seen and admitted to multiple hospitals across Abu Dhabi, including our own, attended different specialists, and underwent a wide range of investigations including blood tests (CBC and differential count, liver and renal profiles and CRP), gastroscopies, colonoscopies, and CT scans and a laparoscopy. The results of all his clinical assessments and investigations did not show any sign.\nOn one of his acute admissions to CCAD, the immunologist was asked to review the patient. Detailed examination of the patient's medical history starting from the onset of symptoms reveled that he used to get 2 swelling episodes every week affecting his face, hands, feet or scrotum and severe abdominal pain twice a week. These swelling episodes and abdominal pain appeared suddenly without any obvious triggering factor, developed over 36 hours and resolved spontaneously in 5-7 days without any medication including analgesics. He reported no laryngeal swellings or respiratory compromise. He denied any fevers, night sweats, weight loss, change in bowel habits or blood in his stools. His swelling episodes were occasionally associated with non-pruritic red skin rash that was mistaken for chronic urticaria for which he was treated with Omalizumab (300mg every 4 weeks) for 9 months without any benefit. Moreover, his response to different types of analgesia, high dose antihistamines, antibiotics and corticosteroids was unsatisfactory. He is a thalassemia carrier; otherwise he is fit and healthy and has no past medical history of note. He has no family history of immunodeficiency, inflammatory bowel disease, autoimmunity or FMF. On examination, he was in pain and his abdomen was soft, tender with guarding and decreased bowel sounds. There was no rebound, rigidity, distension or ascites. He had no peripheral swellings.\nDuring his acute admission to CCAD, a review of his blood tests was performed that showed reduced C4 and absent C1 inhibitor function (Table). These tests were performed a month prior to his admission but were not followed up. His abdominal CT scan showed diffuse swelling and long segment of enhancing mucosal thickening involving the proximal jejunum and gastric mucosa with minimal free abdominal fluid (). In addition, he had a colonoscopy which showed severe mucosal edema in the transverse colon with occlusion of the lumen (). A provisional diagnosis of HAE was made based on his limited complement studies. As he had severe abdominal pain for 24 hours prior to his hospital admission, C1 inhibitor concentrate (2000 units IV over 10 minutes) was administered and within 2 hours his pain had almost resolved.\nAfter his recovery, he underwent detailed immunological investigations that revealed markedly reduced C4 level and absent C1 inhibitor function (performed manually, read on Shimadzu UV-1700 equipment) with normal C3 and C1q levels and raised C1 inhibitor serum levels (). His ENA, total immunoglobulin, CBC and differential count, serum protein electrophoresis, liver function tests, hepatitis serology, lipase, amylase, tissue transglutaminase, stools tests and urinalysis did not show any significant abnormality.\nHe was diagnosed with type 2 HAE based on his abnormal complement studies () and was commenced on tranexamic acid for 3 months. He did not want to start with attenuated androgens because he was concerned about their adverse effects. However, he continued to get abdominal pain even when the dose of tranexamic acid was increased to 3 grams daily. He was then switched to danazol 100 mg twice daily with complete resolution of his abdominal symptoms. |
A 34-year-old female with no significant past medical history presented to our clinic after experiencing a left second metatarsal stress fracture (Figure ). One year prior, while running errands around town, she suddenly felt a sharp pain in her left midfoot and promptly consulted an orthopedic surgeon who placed her in a boot. Six months later, after experiencing minimal improvement in her pain, a different orthopedic surgeon performed an open reduction and internal fixation by injecting 1 mL of bone cement into the diaphysis of the second metatarsal.\nOver the next six months, she noticed no meaningful improvement in her pain. At this point, she presented to our clinic for a third opinion. During our initial visit with her, she stated that her left foot felt different than her right at baseline.\nOn physical exam, there was no gross deformity of her left lower extremity. The skin was intact with a healed incision over the dorsal midfoot, and there was point tenderness to palpation over the second metatarsal. Active and passive range of motion of the ankle and transverse tarsal joint was full and painless. Strength was 5/5 in dorsiflexion, plantarflexion, inversion, and eversion. Sensation to light touch was intact, Achilles reflex was present, and dorsalis pedis and posterior tibialis pulses were palpable.\nLaboratory work revealed an elevated erythrocyte sedimentation rate of 36 (reference range: 0–20) and C-reactive protein of 34.74 (reference range: 0–10.9). Plain radiographs and a computed tomography (CT) scan of the left foot showed diffuse sclerotic changes and cement within the left second metatarsal (Figures , ). Magnetic resonance imaging (MRI) showed diffuse edema of the left second metatarsal with a non-displaced fracture line (Figure ).\nAll treatment options were discussed with the patient and she agreed with undergoing operative fixation. In the operating room, cultures and a bone biopsy of the left second metatarsal were taken. After performing an osteotomy, curettage was performed to remove the injected cement. Open reduction and internal fixation was performed utilizing a plate and calcaneal bone graft (Figure ). The patient was discharged home on the same day with adequate pain control and a bone stimulator. X-rays taken at two weeks post-revision surgery are shown in Figure .\nAt one-month follow-up, her incision was healing well without signs of infection and she had no complaints of pain. At her most recent appointment—three months post-revision surgery—she again reported no pain and good functional recovery with physical therapy. CT scan at three months post-revision surgery showed appropriate alignment of the healing second metatarsal with intact hardware (Figure ). |
A 20-year-old woman presented with a 2-week history of the presence of a painless mass on the left side of the thyroid bed, on which a lobectomy was performed 4 years before. In 2014, this patient arrived at a local hospital with a painless mass on the left side of the neck. Ultrasound disclosed that there was a nodule, which was approximately 3 cm*2 cm in size in the left lobe of her thyroid. Serum thyroid-stimulating hormone (TSH) levels were normal. Lobectomy was performed to treat the nodule, which was diagnosed as GCT following pathological examination of the postoperative tissue. Immunohistochemical results showed that the lesion tested positive for neuron-specific enolase (NSE), S-100 protein and negative for smooth muscle actin (SMA) and thyroglobulin (Tg). The mitotic index was assessed using Ki-67 staining and was found to be below 1%. There were no postoperative complications, such as hoarseness and hypoparathyroidism that were reported after initial surgery. There was no regular follow-up after the surgery was performed.\nThe patient arrived at our hospital after 4 years, in 2018, for the first time after the initial surgery. This time, ultrasound examination revealed a neoplasm, which was palpable through physical examination, on the left thyroid bed, which was considered as recurrence (Fig. a), and multiple solid hypoechoic components of partially cystic nodules and benign lesions were found on the right side of the thyroid. Neck computed tomography scanning with intravenous contrast showed that the mass had a blurred boundary and was slightly unevenly enhanced (Fig. b). The laryngoscopy indicated that bilateral vocal cord mobility was normal. The bronchoscopy showed that a posterior subglottic mass, with normal overlying mucosa, compressed the fibromuscular membrane from the outside (Fig. c). Moreover, cytology of the fine-needle aspiration (FNA) biopsy together with previous surgical history indicated that the current neoplasm was a recurrent GCT of the thyroid.\nTo avoid adding an additional superficial scar, the same anterior approach used during the first intervention was used. Intraoperative neuromonitoring was used to identify and protect the recurrent laryngeal nerve (RLN) and the external branch of the superior laryngeal nerve (EBSLN). Intraoperative exploration showed that the recurrent lesion was about 2 cm in diameter and located next to, and also adhered firmly to the left inferior pharyngeal constrictor muscle (upper side of the thyroid) and to the left RLN at the entrance of the larynx. Using a lancet, the tumor was sharply separated from the left RLN which was completely conserved. The left RLN was not bifurcated before it entered the larynx. The function of the RLN and the vagus nerve (in the cervical sheath) were confirmed to be normal using a threshold (100uV) at the beginning and the end of the operation. The tumor was yellowish in appearance, smooth and rubbery to the touch. Part of the tumor was located behind the fibromuscular membrane and was invasive beyond the midline. This patient accepted surgical dissection along with complete thyroidectomy. After reoperation, this patient did not show signs of postoperative dysphonia and had no clinical evidence of hypoparathyroidism.\nHistopathological examination using H&E staining revealed that the tumor was composed of epithelioid cells with an abundance of eosinophilic granular cytoplasm. The arrow showed that the tumor tissue infiltrated into striated muscle tissue (Fig. a). There was no evidence for nerve invasion in H&E staining. The tumor cells were strongly positive for S-100 protein (Fig. b) and negative for Desmin (Fig. c). Based on the above findings and the patient’s surgical history, post-operative pathology defined the lesion as recurrent benign GCT of the thyroid. After 6 months of follow-up, no local or distant recurrence was found. |
A 30-year-old helmeted male presented to our trauma center following a high-speed motorcycle collision. He was ejected and landed twenty-five feet from his motorcycle. On arrival, he had a systolic blood pressure of 80 mmHg. A right-sided chest tube was placed for clinically diagnosed tension pneumothorax, with significant air but minimal blood returned. Focused assessment with sonography in trauma (FAST) exam was negative for intra-abdominal fluid/bleeding. With resuscitation efforts the patient’s hemodynamics improved. On secondary examination the patient’s right chest wall and shoulder were swollen. The radial pulse on the right was significantly reduced compared to the left. Due to high suspicion for an underlying vascular injury and his improvement in hemodynamics the patient was taken to the interventional radiology suite for imaging. Angiography revealed complete avulsion of the right subclavian artery (Figs. & ). An attempt at endovascular repair was made, however, the guide-wire could not be bridged across the avulsion. A balloon catheter was deployed at the proximal end of the subclavian artery, providing more secure temporary hemorrhage control (Figs. & ). The patient was taken to the operating suite for open exploration and repair of the right subclavian artery. A delto-pectoral approach was utilized. Dissection revealed that primary anastomosis was not possible due to the destruction of the proximal subclavian. Proximal and distal control was obtained with ligation and then the balloon occlusion was removed. Further dissection also revealed complete avulsion with thrombosis of the subclavian vein, which was also ligated. A right common carotid artery to axillary artery bypass was performed using a cryopreserved saphenous vein allograft (CryoLife, Inc. Kennesaw, GA) (). To mitigate the risk of thrombosis formation, intraoperative heparin was used for the duration of the surgery at 500 units per hour. After completion of the anastomosis, angiography of the right upper extremity revealed uninterrupted flow without defect, confirming patency of the vein graft and successful re-vascularization. The patient was taken to the intensive care unit (ICU) in critical condition.\nHe improved in the ICU and resolved his shock state. The following day, subcutaneous enoxaparin for venous thromboembolism prophylaxis was initiated and he was started on dual antiplatelet therapy with oral aspirin and clopidogrel for graft health and the heparin drip was discontinued. He continued to have a strong pulse in both upper extremities. He was discharged in stable condition and follow up at 2, 4 and 12 weeks revealed a patent bypass. This work has been reported in line with the SCARE criteria []. |
A 66-year-old Caucasian woman presented with vertical double vision for the previous 3 weeks. Diplopia was binocular and noticed after she banged her head against the shower screen. She was healthy and had no symptoms suggestive of giant cell arteritis (GCA). A clinical examination showed right fourth cranial nerve palsy. Her magnetic resonance imaging (MRI) scan was normal. Her erythrocyte sedimentation rate (ESR) was 77mm/hour and C-reactive protein 68mg/L. She was commenced on oral steroids while awaiting temporal artery biopsy (TAB) to rule out GCA. TAB revealed normal histology. As there was no clinical suspicion of GCA, her steroids were stopped. The medics investigated her further for raised inflammatory parameters and no cause was identified.\nShe then sought medical attention for a breast lump that she had noticed for the past few months. This was clinically diagnosed as breast cancer. She subsequently underwent right wide local excision of the mass and axillary clearance. Histology revealed 28mm grade 3 oestrogen receptor positive, human epidermal growth factor receptor 2 negative ductal carcinoma involving 12 of 14 axillary nodes. An oncology referral was made.\nMeanwhile, she complained of a droopy right eyelid. Orthoptic examination showed pupil-sparing third nerve paresis and mild sixth nerve involvement. The fourth nerve paresis had resolved. Neuroimaging to rule out cavernous sinus pathology was requested. A MRI scan with contrast showed meningeal thickening in frontal, parietal and occipital lobes and no abnormality in the cavernous sinus territory. This was consistent with carcinomatous meningitis (Figure ).\nBy the time of her oncology appointment, she had developed numbness in the ophthalmic division of her right trigeminal nerve. A bone scan showed bone metastases; computed tomography revealed bone spread and abnormal mediastinal lymphadenopathy. Cerebrospinal fluid (CSF) cytology was negative but protein was slightly raised. Palliative hormone therapy was commenced for metastatic breast cancer. She responded quickly with complete resolution of diplopia, ptosis and numbness on her right anterior scalp within 8 weeks of starting anastrozole. |
A 24-year-old female sustained an injury, while pillion riding a bike, and she was hit and run over by a tractor over both lower limbs and was brought to the trauma center 5 hours after the injury. The patient was normotensive with a blood pressure of 110/80 mmHg and a pulse of 90/minute in the emergency department. The right thigh and knee were swollen and tender with absent anterior tibial artery (ATA) and posterior tibial artery (PTA) without any knee instability ( and ). Capillary refill time and pinprick were delayed. The limb was cold, associated with diminished toe and ankle movements, and diminished sensation over the foot. The left lower limb was shorter with crepitus and abnormal movement in the left thigh and intact distal pulses. Radiographs were performed, which showed left femoral shaft fracture and no bony injury on the right lower limb. Radiographs also showed abnormal soft tissue shadow in the medial aspect of the right distal thigh (). Doppler showed absent ATA and PTA on the right side following which computed tomography (CT) angiogram was performed, which showed non-opacification of right distal superficial femoral artery (SFA) (length of 6 cm) () with popliteal artery reformation, non-opacification of ATA and opacification of PTA and peroneal artery.\nThe patient was taken up for surgery, and the SFA was explored, which was found to be transected with a 7 cm contused segment of the vessel around 10 cm proximal to the knee joint. The contused segment was resected, fogartisation of distal and proximal ends was done, and reconstruction with reversed saphenous vein interposition graft was undertaken using 5-0 prolene. Medial and lateral fasciotomy for right leg was conducted and external fixator was applied for left femur fracture.\nPostoperatively, heparin and low-dose aspirin were given. The limb was warm with normal capillary refill, and Doppler showed biphasic flow in ATA and PTA. Fasciotomy wound was treated with vacuum-assisted closure and removal of the fixator and internal fixation using closed intramedullary nail (), and primary closure of fasciotomy wound was performed after two weeks. Follow up of patient after one month was uneventful with palpable clinical pulses and normal sensation and movement in her right lower limb. |
A 64-year-old man who had undergone coronary artery bypass grafting (LIMA to LAD) and AVR in 2000. Afterwards, a cardiac pace maker was implanted because of 3rd degree atrioventricular block. He had diabetes and hypertension. He underwent coronary angiography in 2010, and he was detected with a branch arising from the proximal segment of the LIMA, which was not occluded during the surgery (). A coil occlusion procedure was applied via three piece of coil (Boston Scientific Corporation; 3 mm x 3 cm) (, ). After this coil occlusion procedure was successfully performed in 2010, he was symptom-free and only consumed warfarin owing to AVR.\nHe was admitted to the coronary intensive care unit with unstable angina after 4 years of the first coil procedure. Hypokinesia was detected on the anterior wall of the left ventricle with transthoracic echocardiography (LVEF, 45%). Coronary angiography revealed that the bypass graft was patent but recanalization was seen on the LIMA (). Myocardial perfusion scintigraphy showed ischemia on the anterolateral wall (). Percutaneous coil reocclusion was attempted on the side branch of the LIMA. Warfarin was ceased before the procedure, and bridging therapy with enoxaparin was initiated. LIMA was selectively cannulated with a left 6-F Judkins catheter; accordingly, a microcatheter was advanced over the guidewire into the side branch. Two coils were released into the appropriate position (Tornado, Cook Medical; 2 mm x 4 cm and 3 mm x 6 cm). During the third coil implantation, an attachment apparatus was detached from the proximal part of the hinge point in the LIMA (Tornado®, Cook Medical; 5 mm x 7 cm) (). We attempted to push the detached part into the side branch via a pusher and catch the detached part of the coil via a micro-snare, but none of our attempts was successful. During the procedure, severe vasospasm was seen on the LIMA, and the patient suffered from angina. Implantation of everolimus-eluting stent (3.0 × 23 mm) into the LIMA next to the unligated side branch was considered with an aim to fix the detached part of the coil (). After the procedure, the flow of the side branch of the LIMA was ceased and the detached part of the coil was trapped between the stent and vessel wall (, ). Warfarin was reinitiated 2 days after the procedure. Following coil occlusion and stenting, ischemia on the anterolateral region disappeared on scintigraphy (). The patient was mobilized without any complication following the procedure. |
A 35-year-old man presented at a local hospital with epigastric pain. Esophagogastroduodenoscopy (EGD) showed that an IVC filter strut had penetrated the third portion of the duodenum (arrow, Fig. ), and this was confirmed by computed tomography (CT) (arrow, Fig. ). In order to retrieve the IVC filter, the patient was referred to our department. He had a history of testicular cancer with para-aorta lymph node metastasis. Left renal vein thrombosis developed because of neoadjuvant chemotherapy before RPLND, and anticoagulants were administered before RPLND. Three years previously, he had undergone left orchiectomy, retro-mediastinal lymph node dissection, and RPLND at the previous hospital. The left common iliac vein was intraoperatively damaged during RPLND. Because the previous surgeon was worried about the high incidence of postoperative DVT and PTE, anticoagulant therapy was continued after RPLND. However, because DVT developed in the left common iliac vein after the initial surgery, a retrievable IVC filter (ALN, France) was placed in the IVC caudal to the renal vein to prevent PTE, and the patient had been receiving anticoagulant therapy. Because follow-up CT after IVC filter placement showed that DVT persisted at the left common iliac vein despite anticoagulant therapy, the IVC filter could not be retrieved at the previous hospital.\nEnhanced CT also revealed that DVT remained in the left common iliac vein. Because ultrasound examination showed organized DVT, an IVC filter was considered unnecessary. An endovascular approach was considered unfeasible for retrieval because two of the filter struts had penetrated the duodenal wall. An extensive discussion with an internal medicine specialist was performed. Because DVT remained with no remarkable changes in CT images for 3 years and the incidence of PTE caused by DVT in the left common iliac vein would be low, we chose surgical treatment for this patient in order to prevent bleeding at the duodenum.\nThe IVC filter was retrieved through cavotomy, and the duodenal penetration site was repaired using intraoperative EGD clipping. The operation lasted 5 h and 54 min, and the intraoperative bleeding volume was 1172 mL. Because it was not possible to mobilize the duodenum due to adhesions resulting from the previous surgery, the IVC at the sites caudal to the renal vein could not be explored. However, a mesenteric incision caudal to the third portion of the duodenum enabled encircling and taping of the IVC (Fig. ). After clamping the IVC cranial and caudal to the duodenum, a 5-cm vertical incision was made on the IVC cranial to the duodenum and the IVC filter was retrieved (Fig. a). Although the head of the IVC filter had penetrated into the IVC intima, we were able to bluntly peel the filter head from the intima. The IVC incision was closed using a continuous 5-0 Prolene suture (arrow, Fig. b). The IVC clamping time was 22 min. Intraoperative EGD revealed no bleeding at the duodenal penetration site (Fig. ). To prevent duodenal bleeding or perforation, the penetration site was repaired by EGD clipping. Retrieved IVC filter was presented as (Fig. ). The patient did not develop any postoperative complications and was discharged on postoperative day 16. |
A 69-year-old man presented a 3-month history of persistent dull pain in the left lower abdomen. The patient experienced occasional diarrhea, slightly black stools, a poor appetite, fatigue, and 20-lb weight loss. There was no nausea, vomiting, fever, or night sweats. The patient denied any personal or family history of cancer. Before being transferred to our hospital, the patient underwent a gastrointestinal endoscopy that showed no abnormalities. Abdominal CT revealed a large, irregular mass in the pelvic cavity () that was considered as a intestine diverticulum with an abscess. He was suspected to have an inflammation and was treated with a two-week course of antibiotics in another medical facility. However, the antibiotics did not relieve his symptoms. His hemoglobin level was 111 g/L (standard 135–170 g/L), and occult blood (OB) test was positive. Biochemical infection screening and tumor markers (CEA, AFP, CA199, CA724, CA125) were all normal on admission. On physical examination, he was found to have a well-defined soft mass on the left lower abdomen, poor mobility, slight tenderness. Ultrasonography revealed a 6.1 3.8 × 4.2 cm irregular, heterogeneous hypoechoic mass in the left lower abdomen (\n). The tumor contained multiple necrotic areas and close contact with the bladder wall. Heterogeneous enhancement, and wash-out time of 54 s on CEUS ().\nThe PET–CT was performed revealing a highly metabolic mass in the lower abdomen and multiple metabolic nodules in the mesentery (). This was considered a small intestinal stromal tumor with lymph node metastasis. The patient did not show symptoms of lymphoma, and no abnormalities were found in other organs. The consensus of the attending radiologists and surgeons was that it was a malignant tumor rather than an inflammatory process. A core needle biopsy was rejected because of the broad area of necrotic tissue; there was also concern that adequate tumor tissue would not be obtained while risking intestinal perforation or tumor dissemination. Finally the patient underwent surgery to remove the lesion. A 5× 6 cm brown cauliflower-like mass was resected from the ileum, 50 cm away from the ileocecal junction and the surrounding lymph nodes. This mass invaded the serosal layer at the inferior portion of the bladder.\nGross pathology showed the tumor to be friable and having multiple ulcerations on the surface (). H&E sections revealed a small, blue, round tumor (). Histopathological examination showed positive CD99, CK (pan), Ki67 (70%+), Fil-1, and CD34 levels (). Molecular analysis revealed positive EWSR1 fusion gene transcripts, as shown by RT-PCR (). Based on morphology and immunohistochemistry, the tumor was diagnosed as EES/PNET. The patient refused to receive chemotherapy after surgery. He was requested to come to the hospital for examination every month for the first half year, but he did not follow the advice. He came to the hospital because a soybean-like mass was on his forehead, and denied any other symptoms. An MRI was performed revealing a 2.3 × 2.1 × 2.3 cm lesion on the right side of the frontal bone (). The mass extended to the skull and invaded the brain tissue. A second surgery was performed to remove the lesion and adjacent erosive bone. Post-operative pathologic diagnosis revealed the same histology, i.e. the mass metastasized from the primary small intestinal tumor. We performed a telephone follow-up with this patient every two months but learned that he did not undergo any further treatment after the second surgery and died 6 months later; there was no more information about his death. |
A 43-year-old man with a history of chronic alcoholism presented with abdominal distension. The previous day, the patient had presented to a local hospital with anal bleeding and abdominal pain after an incidental insertion of barbecue skewer per anus in the drunken state; subsequently, he had undergone sigmoid loop colostomy for rectal perforation. However, after the operation, the patient had become hemodynamically unstable. At presentation, his systolic blood pressure was 90 mmHg and the pulse rate was 135 beats/min. Although there was no gross rectal bleeding, the digital rectal examination revealed a penny-sized anterior rectal wall defect 6 cm from the anal verge (AV). Computed tomography (CT) revealed a hematoma (12 × 10 × 15 cm) with active bleeding in the pelvic cavity and a pseudoaneurysm in the anterior wall of the rectum (). Since the patient was hemodynamically unstable, an emergency operation was performed. During the operation, a massive subperitoneal hematoma in the rectovesical pouch and large amount of blood in the peritoneal cavity were found. After evacuation of the hematoma and blood, oozing continued in the rectovesical pouch (). Thus, compression with gauze was performed for 30 min until the oozing stopped. The Hartmann procedure was performed with the suspected bleeding focus included, but the perforation site was not included.\nAlthough the postoperative course was uneventful and there was no evidence of recurrent bleeding on the follow-up CT on the 7th postoperative day (POD), a focal enhancing lesion in the anterior wall of the rectum indicating a residual pseudoaneurysm was noted (). On the 11th day POD, his hemoglobin decreased from 11.6 g/dL to 7.9 g/dL, and the follow-up CT revealed recurrent hematoma (6.0 × 4.2 cm) in the pelvic cavity and the residual pseudoaneurysm (). Following the diagnosis of recurrent bleeding from the residual pseudoaneurysm, an angiography was performed. However, the angiography failed to localize the pseudoaneurysm, and definite signs of extravasation could not be ascertained. Thus, prophylactic gelfoam embolization at the anterior branch of both the internal iliac arteries was performed (). The subsequent hospital course was uneventful, and the patient was discharged on the 25th POD. After 3 months, the previous rectal lesion (AV: 6 cm) healed, and colostomy reversal was performed without morbidity. |
Abdominal computed tomography for regular monitoring of uterine fibrosis in a 49-year-old woman found a hypodense lesion 7 cm in diameter with a clear boundary near the gallbladder (Fig. a, b). No obvious change in size and internal density of uterine fibrosis had been observed during these 5 years (Fig. b). She had no previous history of alcohol or drug abuse. Ultrasound revealed a well-defined, non-calcified tumor between the gallbladder and liver (Fig. ). Magnetic resonance imaging demonstrated a hypointense tumor that compressed the gallbladder and liver on precontrast T1 mapping (Fig. a). T2-weighted images revealed hyperintense tumor with delayed enhancement on arterial phase and portal venous phase (Fig. b) followed by a delayed washout on the hepatocyte phase. Clinical evaluation and laboratory results were nonspecific, and serum tumor markers including carcinoembryonic antigen, cancer antigen (CA) 19-9, alpha-fetoprotein (AFP) and squamous cell carcinoma antigen were within their normal ranges. The findings were consistent with a gastrointestinal stromal tumor (GIST) or another benign tumor originating from the liver or gallbladder. Accordingly, we planned firstly laparoscopic partial hepatectomy concomitant with gallbladder resection. Lymphadenectomy among the hepatoduodenal ligament were also planned under the situations where malignant tumor was suspected intraoperatively. Under the laparoscopic view, a soft tumor had expansively progressed behind the gallbladder, with dorsal compression of the liver (Fig. ). The tumor was easily separated from Laennec’s capsule of the liver at the gallbladder neck and body without adhesion, which indicates the tumor is benign. Therefore, operative policy changed to tumor enucleation with cholecystectomy. Frozen sections including the surgical margin of the cystic duct were negative for tumor cells. As the tumor was moderately attached to the liver at the fundus of the gallbladder, the liver parenchyma was partially resected en bloc to ensure that the surgical margins were free of tumor tissue.\nThe resected specimen included soft and white tumor tissue with clear boundaries that was located between the liver and gallbladder (Fig. ). Pathological examination following Hematoxylin and Eosin and immunohistochemical staining of tumor specimens was consistent with SFT. The specimens included spindle-shaped tumor cells with elongated nuclei (Fig. a), present in both tangled and patterned arrangements in the subserosal layer of the gallbladder (Fig. b, c). The tumor tissue had a storiform pattern with alternating hypocellular and hypercellular areas with some showing myxoid degeneration. The tumor cells were positive for CD34, CD99 and B-cell lymphoma (BCL)-2 and negative for S100 and alpha smooth muscle antigen (αSMA) staining (Fig. d), and few cells were positive for the cell proliferation marker Ki-67/MIB-1. The tumor cells were negative for SMA, keratin, cytokeratin (AE1/AE3), CD117, epithelial membrane antigen, and desmin. The pathological diagnosis was SFT originating from the cystic plate.\nThe postoperative course was uneventful. She was discharged on postoperative day 9. Chemotherapy was not considered necessary, and postoperative follow up at 18 months found the patient well without any sign of recurrence. Periodical check-up for uterine fibrosis by gynecologist is also continued. |
A 71-year-old Caucasian woman with symptomatic kyphoscoliosis and multilevel lumbar spinal stenosis had been followed up in our clinic for sagittal imbalance and fatigue with progressive low-back and leg pain as well as subjective weakness of the legs bilaterally (). Physical therapy and epidural steroid injections failed to improve her symptoms. Her strength and reflexes were normal in the lower extremities. She was elected for surgery after conservative measures failed to improve her symptoms.\nWe performed T10 to pelvic fixation with posterior laminectomies from L2 to S1, a pedicle subtraction osteotomy at L3 to correct her sagittal imbalance, and transforaminal lumbar interbody fusion at L5-S1 to decrease the L5-S1 pseudarthrosis rate. Initial neurophysiologic monitoring demonstrated consistently robust responses. An osteotome and mallet were used to resect the IAPs in preparation for pedicle screw placement.\nDuring resection of the leftT11 IAP, somatosensory evoked potentials (SSEPs) and motor evoked potentials (MEPs) were lost immediately after impact of the osteotome. The resection of this IAP was performed in a standard fashion and the spinal canal was not violated during this maneuver. We further inspected the area and did not find fracture of the adjacent superior articular process or lamina. The screw tracts were checked and none of the pedicle screws violated the spinal canal. We reviewed her preoperative MRI and the canal did not demonstrate clear stenosis or compression at that level (). In an attempt to restore monitoring, her mean arterial pressure (MAP) was increased and sedation decreased. We contemplated performing a Stagnara test, but the SSEPs and MEPs improved. Given this improvement, and the difficulty in performing a Stagnara test in a geriatric patient, we elected to proceed with the operation. During closure of the PSO, the MAP was maintained above 90 and the osteotomy closed well with only minor dural buckling. The operation was further complicated by an unintended durotomy at the L3-4 level which was primarily closed.\nPostoperatively, the left leg was plegic with 2/5 strength in the right leg. While still intubated, we transported her to the computed tomography (CT) scanner. Thoracic CT scans demonstrated a preexisting area of hypertrophic bone not obvious on the MRI which was reviewed intraoperatively (). Bilateral osteophytes, consistent with OLF, were occupying the canal at the T10-11 level. The osteophyte on the left was larger than the one on the right. We felt the compression secondary to the OLF was likely aggravated by the use of the osteotome during the SPO. Therefore, we returned to the OR for emergent decompression. This was achieved with a T10-11 laminectomy and bilateral OLF resection with the M8 drill and Kerrison instruments (). After the laminectomy, strength was 4/5 in the right leg, 2/5 in the proximal left leg, and 4/5 in the distal left leg.\nHer postoperative course was complicated by a pulmonary embolism on postoperative day (POD) three, which required systemic anticoagulation. Her neurologic exam improved minimally prior to discharge on POD 10, and she was still unable to independently ambulate at that time. On POD 15, the patient returned with cerebral spinal fluid (CSF) leaking through the caudal aspect of the incision. She required two additional operations and temporary CSF diversion via a lumbar subarachnoid drain to stop the leak. By POD 32, the incision was fully healed and she had no residual signs or symptoms of persistent CSF leakage. Her postoperative course was further complicated by a non-ST elevation myocardial infarction treated with aspirin and beta blockers. She was discharged to a rehabilitation facility on POD 40.\nTwo months postoperatively, she had full strength and was able to walk independently. However, she had some residual lower extremity hyperreflexia secondary to the intraoperative spinal cord injury. She reported that while her preoperative symptoms were not completely resolved, they were improved. She continues to ambulate independently upon one-year follow-up, with no radiographic evidence of hardware failure (). |
An 80-year-old gentleman with recently diagnosed metastatic bladder cancer to bones and retroperitoneal lymph nodes was started on pembrolizumab. Patient was not considered for cisplatin-based therapy given decreased performance status and chronic renal impairment. Patient did have a history of lupus treated with mycophenolate and hydroxychloroquine. Given his clinical condition and comorbidities, his oncologist considered pembrolizumab is a better option for the patient as his lupus is well controlled. He presented to the emergency room with worsening weakness and inability to walk after receiving his first cycle of pembrolizumab. He has been deconditioned over the past few weeks but was able to walk with a walker and perform daily activities at baseline. However, he was unable to get up from the bed and was extremely weak in bilateral lower extremities after receiving immunotherapy and hence sought medical attention.\nHe reported extensive past medical and surgical history that includes hypertension, hyperlipidemia, hypothyroidism, lupus, peripheral arterial disease status post stent placement in right mid popliteal artery, coronary artery disease status post percutaneous intervention, tachy brady syndrome status post pacemaker placement, congestive heart failure with ejection fraction of 40%, chronic obstructive pulmonary disease, squamous cell carcinoma of supra-glottis status post radiation therapy in remission, chronic back pain, and benign prostatic hypertrophy.\nHe is a former smoker, quits smoking 10 years ago, drinks alcohol occasionally, and reports no illicit drug use. There is no significant family history. Medication list includes apixaban, clopidogrel, levothyroxine, mycophenolate, atorvastatin, hydroxychloroquine, tamsulosin, finasteride, and omeprazole. On initial examination, his vitals are stable. Physical examination was significant for the patient in distress from back pain radiating to bilateral lower extremities associated with weakness. Also noted chronic macular darkening of leg. Neurological examination was significant for weakness in the lower extremities with a strength of 3 out of 5 bilaterally. There was no sensory loss. The patient did have a normal rectal tone. The rest of the physical examination was unremarkable. Patient’s urinalysis was negative. Portable chest x-ray revealed emphysema, no other acute findings. Patient’s admission labs are summarized in .\nHospital course: Due to lower back pain and weakness in the lower extremities, he underwent computed tomography (CT) of the cervical, thoracic and lumbar spine which revealed multiple bony sclerotic lesions consistent with metastasis and multifocal lymphadenopathy. The patient received 10 mg of dexamethasone initially. Magnetic resonance imaging (MRI) was not done due to pacemaker placement 2 months ago. Neurosurgery recommended no indication for surgical decompression. With a history of Peripheral arterial disease and associated chronic skin discoloration in lower extremities, arterial ultrasound was ordered, which showed occlusion of left distal superficial femoral and left dorsalis pedis arteries with extensive plaque formation bilaterally. Vascular surgery was consulted who reviewed the ankle-brachial pressure index (ABPI), which is more specific for lower extremity ischemia, which was negative. They concluded that the weakness is unrelated to arterial insufficiency.\nOncology team presumed that it could be lupus flare related to immunotherapy and recommended checking antinuclear antibody (ANA), rheumatoid factor (RF), anti-cyclic citrullinated peptide (CCP), complement levels. The results came back as ANA positive confirming lupus however had negative complement levels and undetectable ds DNA negating acute flare. Neurology was consulted due to worsening weakness, after the pacemaker representative was contacted MRI of cervical thoracic and lumbar spine was obtained which revealed multilevel stenosis, small C3 sclerotic lesion, no definitive fractures, or metastasis. The imaging studies are summarized in . The thyroid-stimulating hormone, 25-hydroxy vitamin D, vitamin B12, and folate levels were all within the normal range. Creatinine phosphokinase (CPK) was checked due to suspicion of medication-induced neuromuscular weakness associated with the use of hydroxychloroquine, which was reported as normal. Additional laboratory findings are summarized in .\nDue to persistent, progressive weakness both in lower and upper extremities, as well as worsening shortness of breath. A more detailed neurological examination was done that showed absence of deep tendon reflexes in lower extremities, downswing plantar reflexes, decreased power in bilateral lower extremities 2/5, decreased handgrip 2/5, and absent vibration sense below the knees. Lumbar puncture was done after holding apixaban for 48 hours and cerebrospinal fluid studies revealed normal cell count, cytology with no evidence of infection. It did show increased protein with increased albumin cytological dissociation confirming Guillain Barre syndrome, which could be related to pembrolizumab. The lumbar puncture findings are summarized in . The patient’s forced vital capacity dropped to 14 mls/kg due to respiratory muscle weakness. The patient was then started on intravenous immunoglobulin daily for 5 days with close monitoring of forced vital capacity. Patient’s forced vital capacity increased to 21mls/kg at the time of discharge. Neurology recommended outpatient electromyography/nerve conduction studies. Oncology determined that he was not a candidate for chemotherapy given his poor functional status and recommended hospice. The patient’s weakness started to improve and was recommended to transfer to a rehabilitation home for continued physical therapy. The patient and his family want to pursue hospice after discharge from the rehabilitation home. |
A 62-year-old man who had 15 years earlier undergone partial resection for left sphenoorbital meningioma in another hospital presented with a 1-year history of progressive proptosis of the left eye. He was referred to our hospital because magnetic resonance imaging (MRI) and computed tomography (CT) revealed a hyperostotic tumor of the left sphenoid wing with orbital and middle fossa extension. On admission, ophthalmic examination showed slight proptosis of the left eye, but visual acuities, visual field areas, eye movements, and pupillary light reflexes remained normal. Neurological examination showed no neurological deficits. The results of laboratory examinations, including thyroid function, were essentially normal. CT performed in our hospital revealed hypertrophy of the left sphenoid wing, orbital lateral wall, and floor of the middle fossa [Figure and ]. MRI showed a Gd-enhancing tumor occupying the left sphenoid wing and orbital lateral wall []. Extension of the tumor into extracranial structures such as the sphenoid sinus, pterygopalatine fossa, and infratemporal fossa as well as adjacent structures such as the cavernous sinus and SOF was also revealed [Figure and ]. Cerebral angiography demonstrated hypervascularity of the tumor supplied by the branches of the internal maxillary artery []. Although detailed information about the previous operation was unavailable, including histopathological examinations and surgical procedures, the radiological findings were compatible with gross extension of recurrent sphenoorbital meningioma. Preoperative embolization using N-butyl cyanoacrylate was performed 1 day before the surgical resection []. Based on MRI findings of tumor extension into the sphenoid sinus from the middle fossa with broad continuity, surgical resection through combined simultaneous transcranial and endoscopic endonasal approaches was planned for aggressive surgical resection with minimized risk of postoperative CSF leakage. Subtotal resection was performed, leaving residual tumor in the cavernous sinus and SOF. Postoperative CT showed extensively drilled skull base bone [Figure –]. Postoperative neurological examination revealed no neurological deficits, including ocular movement and facial sensation. A lumbar spinal drain was placed for 5 days postoperatively and no CSF rhinorrhea developed. Histopathological examination identified the tumor as meningothelial meningioma, World Health Organization grade I. Follow-up MRI at 3 months postoperatively demonstrated residual tumor only in the cavernous sinus and SOF [Figure –].\nUnder general anesthesia, the patient was placed supine with the head fixed in a Mayfield clamp and rotated 30° to the right. To achieve both transcranial and endoscopic endonasal approaches, two surgical teams worked simultaneously. The transcranial surgeon stood at the head end of the patient and the endonasal surgeon stood on the right side of the patient.\nThe frontotemporal craniotomy made 15 years earlier was used and an additional zygomatic osteotomy was created to approach the tumor. The thickened hyperostotic sphenoid wing and lateral wall of the orbit were completely drilled out and removed. After cutting of the meningo-orbital band followed by peeling of the dura propria of the middle fossa from the lateral wall of the cavernous sinus, further drilling around the SOF and floor of the middle fossa was performed. Following identification of the maxillary and mandibular nerves, the floor of the middle fossa around the foramen rotundum and foramen ovale was drilled until the pterygopalatine fossa and infratemporal fossa were exposed. During these procedures, the tumor mass extending into the sphenoid sinus was exposed and the sphenoid sinus was then widely opened by resection of the tumor mass []. All skull base bone infiltrated by the tumor was extensively drilled out and removed. Tumor-involved dura was resected and repaired with a free graft of fascia lata. Tumor extension into the cavernous sinus and SOF was left in place to avoid the risk of postoperative ocular nerve palsies. The large bone defect of the sphenoid sinus was covered with a free graft of fascia lata and then fixed with fibrin glue from the transcranial side [].\nA rigid high-definition endoscope (Karl Storz, Tuttlingen, Germany) was used in a standard endonasal approach to expose the sphenoid sinus. A pedicled NSF was then prepared while maintaining the vascular supply from the posterior septal branch of the sphenopalatine artery.[] Because the tumor had expanded laterally along with development of the lateral recess of sphenoid sinus beyond the sphenoid body into the greater wing [], the transmaxillary transpterygoid approach was employed to approach the tumor extending laterally over the vidian canal.[] Uncinectomy, ethmoidectomy, and sphenoidotomy were performed in a stepwise manner. After the vidian nerve and artery were coagulated and sectioned, the contents of the pterygopalatine fossa were subperiosteally dissected and retracted laterally enough to expose the foramen rotundum. Tumor expanding laterally in the sphenoid sinus was then removed en bloc []. After resection of the tumor from the transcranial approach, the large skull base defect between the middle fossa and sphenoid sinus was covered with a pedicled NSF and then fixed with fibrin glue []. The NSF was secured with placed Surgicel®, then buttressed by gauze packing in the sphenoid sinus. |
A 5-year-old previously healthy, VZV unvaccinated boy presented to our emergency department with typical varicella skin lesions which had developed 2 days prior. He had a history of fever and poor oral intake. Furthermore, he complained of pain around the left thigh and was reluctant to bear weight. The child was in a mildly reduced general condition with normal heart rate, respiratory rate and blood pressure for age. He was febrile with a temperature of 39.6°C. The cardiopulmonary examination was unremarkable. Next to multiple crusted skin lesions there was a tender and discolored area (3–5 cm) on the left buttock (). The boy refused to sit or lie on his back.\nLaboratory work-up showed a white blood cell count (WBC) of 7.2 G/L, platelets of 131 G/L and a CRP of 195 mg/L. Blood cultures were drawn and intravenous Cefuroxime and Clindamycin were started for suspected bacterial soft tissue infection. Growth of GAS from the blood culture was reported with a time to positivity of 2.6 h by the microbiology laboratory. Ultrasound showed signs of soft tissue inflammation around the painful area at the buttock. On the second day of hospitalization the patient had progressively worsening pain of the left thigh. A CT scan revealed inflammation and swelling of the gluteal muscle. Urgent surgical debridement was performed and intraoperatively necrotising fasciitis was confirmed. Tissue swabs grew GAS. Although antimicrobial treatment was started promptly, GAS was still detected in the tissue samples 48 h after initiating betalactam and lincosamide antibiotics at the first debridement. Further blood cultures were not taken at this time. As there was little improvement during the following days, an MRI was performed showing multiple abscesses in the gluteal muscle but no osseous involvement. Overall the child needed two further debridements on days 3 and 4 of hospitalization with application of a vacuum assisted closure (V.A.C.) therapy. On day 5 of hospitalization the patient presented with respiratory distress and required supplementary oxygen. He was transferred to the Pediatric Intensive Care Unit (PICU). On clinical examination a new systolic murmur was heard. Echocardiography revealed mitral valve prolapse with regurgitation. Assuming an endovascular infectious complication, a further set of blood cultures was drawn (which remained sterile) and antibiotic treatment was changed empirically to Gentamycin and Ceftriaxone. Two days later the boy's general condition deteriorated further and a second echocardiography revealed progressive prolapse of the mitral valve, assuming rupture of the chordae tendineae (). X-ray of the chest revealed pulmonary infiltrations due to mitral regurgitation (). The child was intubated and transferred to a tertiary pediatric cardiac surgery center where the mitral valve was reconstructed the next day and neo-chordae were implanted. Endocarditis was confirmed intraoperatively (small proliferative inflammatory changes of the endocardial tissue) and antibiotic treatment was adjusted to intravenous amoxicillin and continued for 4 weeks. At the day of transfer to the cardiac surgery tertiary center, CRP was 32 mg/l, WBC 15.9 G/L and the child was afebrile. The last documented laboratory findings after 4 weeks of antibiotic treatment showed a CRP <4 and WBC 3.97 G/L and a blood sedimentation rate of 28 mm/h. The wound on the buttock was successfully closed 2 weeks after placement of the V.A.C. Four and a half weeks after primary admission the patient was discharged home in good clinical condition. Cardiology follow-up 1 month later revealed good biventricular function and only mild mitral regurgitation. Screening investigations for an underlying immunodeficiency (quantitative and qualitative humoral and cellular testing and HIV screen) were unremarkable. S. pyogenes M serotyping was not done by our laboratory. |
A 72-year-old man was referred for resection of a cecal mass that was found on routine colonoscopy (Fig. ). The patient denied history of blood per rectum, change in bowel habits, abdominal pain, nausea or weight loss. The patient had a significant past medical history for prostate cancer for which he underwent prostatectomy and chemo-radiation therapy. He also underwent open repair of bilateral inguinal hernias in the remote past; the operative notes were not available to us. Tumor markers (CEA and CA19-9) and routine blood tests were unremarkable.\nThe mass was found in the cecum during routine colonoscopy (Fig. ); it had raised borders, did not have a stalk and was not associated with mucosal erythema or ulceration. Biopsies showed normal colonic mucosa. A computed tomography (CT) scan of the abdomen showed a 3.3 × 4 cm mass that compressed the wall of the cecum. There was no associated lymphandenopathy or liver lesions (Fig. ). Based on these findings, we could not conclude if the aforementioned mass was intramural or extramural to the cecum. Due to the uncertainty about the location of this mass (intramural versus extramural) and the inability to rule out a neoplasm, we proceeded with a diagnostic laparoscopy with the possibility of resection of the cecum. Intraoperatively, we found that the right inguinal hernia had been repaired using a plug-and-patch technique. The mesh plug was fixed in the preperitoneal space and had not violated the peritoneal layer. The ‘Plug’ had not fully deployed in the preperitoneal space and had retained a cone-like shape. The wall of the cecum appeared normal and there were no masses in its anterior wall. An intraoperative colonoscopy was done to confirm the absence of a mass; upon insufflation of the colon, it became apparent that the cecal wall was pressed against the non-deployed plug thereby creating the impression of a mass with extrinsic compression. |
A 20-year-old man presented with multiple lobulated growths of the right lower limb, lymphorrhea and disparity in limb girth. In the first 12 years of life, the limb was proportionate. Thereafter, for next 8 years, the right lower limb overgrew progressively which was painless. There were intermittent episodes of infection which subsided with antibiotic therapy and local care. The patient became physically and socially handicapped and lost his job. On inspection, the right lower limb below knee and dorsum of foot had multiple lobulated masses mimicking grade IV lymphoedema. The skin over the swelling was rugouse with dark pigmentation. The sole was spared. There was another lesion in the upper thigh extending to the groin. The lower two-third of the thigh was spared [Figure –].\nOn palpation, it was soft, non-tender, compressible and non-pulsatile []. There was a significant reduction in size on elevating the limb. On compression, we could see and feel the fluid gushing towards a pouch in the lower thigh region. Again on lowering the limb, the fluid could be felt rushing back to below knee region – a peculiar feeling []. Aspiration yielded an amber-colored fluid.\nThe differential diagnoses in our mind were lymphoedema, lymphangioma, lymphangiomatosis and lymphangiectasis. The patient was subjected to detailed investigation. The MRI of lower limbs and pelvis showed an extensive mass involving predominantly the subcutaneous tissue of right thigh with retroperitoneal extension. There was encasement of femoral and iliac vessels []. MR angiography showed narrowing of right iliac and femoral vessels due to extrinsic compression. There was no intrinsic vessel involvement []. The colour Doppler study showed normal patency of the limb vessels with normal flow []. Final diagnosis was established by lymphangiography and lymphoscintigraphy which revealed marked dilatation of lymphatic channels in leg and inguinal region []. The cytology of the aspirated fluid showed mature lymphocytes in a protein-rich background. Thus, the diagnosis of lymphangiectasis was confirmed.\nA two-staged excision was planned for the leg and thigh separately. The below knee lesions were excised from the dorsum of foot, ankle and pretibial region by three individual incisions. Soon after incision, straw-coloured lymph started flowing freely. About 2 liters of fluid was completely squeezed out. There was hardly any delineation of subcutaneous tissue from dermis to deep fascia. Every structure was replaced by a dense, dilated network of lymphatic channels [Figure ,]. It was excised in full depth along with a portion of excess skin. The wound was closed primarily, and pressure dressing was applied. Stitches were removed on 12th day. After 6 months, the groin lesion was excised. No attempt was made to excise the retroperitoneal mass.\nThe histopathology of the excised specimen confirmed it to be lymphatic tissue with interconnected, dilated lymphatic spaces []. On regular follow up, there was no recurrence after 3 years. The result was excellent although hyperpigmentation of the skin persisted []. |
A 73-year-old Caucasian male with past medical history of Pott's disease as a child, osteoarthritis of the right knee, and a right Baker's cyst presented to the Emergency department with complaints of worsening leg pain for two days and confusion for one day. At presentation, the patient was tachycardic. All other vital signs were within normal limits. His review of systems was remarkable for chronic knee pain for the last two years and right leg pain for the last three months and decreased appetite and weight loss over the course of four months. On physical examination, the patient was in moderate distress. He was alert and oriented to time, place, and person but had poor concentration. On musculoskeletal exam there was a diffuse area of swelling on the right lower extremity which was extremely tender to touch with minimal warmth and erythema. This was located posteromedially involving the calf and medial aspect of the leg. Knee joint was nonswollen, nontender, and without any erythema or warmth (). Laboratory data revealed leucocyte count 14.6 k/cumm, Hemoglobin 10.2 mg/dL, and hematocrit 30.4%. HIV testing was negative. His metabolic panel showed hyponatremia, 124 mEq, but was otherwise unremarkable. Leg MRI was performed. This showed a large heterogeneous fluid collection involving the subcutaneous soft tissues of the posterior medial right lower extremity measuring approximately 21.1 × 7.2 × 8.5 cm with marked mass effect on the gastrocnemius muscle. Heterogeneity of the distal femur including the femoral condyles, tibial plateau, and proximal fibular head was also visualized suggesting possible osteomyelitis as well (). The patient underwent surgical resection of the left Baker's cyst. Intraoperative findings included a thick, boggy synovium with dull and dusky appearing cartilage. Tissue and fluid stain revealed acid fast bacillus (AFB) in one of two samples which was later identified as MTB. On pathologic section, a granulomatous reaction with giant cells was present. The patient was placed in airborne isolation and a workup for dissemination was pursued. Respiratory samples also had AFB identified on initial acid fast staining. Chest CT showed multiple apical cavitary lesions and nodular infiltrates with tree in bud opacities in the apices and right lower lobe. Acute kyphotic deformity of the upper thoracic spine with loss of vertebral body height, vertebral body fusion, posterior element fusion, and kyphosis consistent with the patient's known history of Potts disease was also seen in chest CT. Due to worsening mental status, a diagnostic lumbar puncture was also performed. CSF analysis showed 1 red blood cell/UL, 151 white blood cells/UL, 64% neutrophils, protein 1668 mg/dL, and glucose of 8 mg/dL. CSF polymerase chain reaction tested positive for MTB. Testing for HIV, Hepatitis B, and C was negative. Treatment with rifampicin, ethambutol, isoniazid, pyrazinamide, and dexamethasone was started. During the course of therapy, patient developed pneumonia and hypoxic respiratory failure requiring intubation. Family chose to proceed with comfort measures only and the patient passed away due to septic shock. |
This case involves a 46-year-old man with a medical history of hypertension, obstructive sleep apnea, and pericarditis who sustained a left ACL tear after a noncontact twisting injury while playing basketball. After a trial of physical therapy and anti-inflammatory medication, he still experienced persistent pain and instability and elected to undergo arthroscopic ACL reconstruction with a peroneus allograft. The graft was fixed to the femur using a suture button and the tibial side was fixed with a tenodesis implant. No complications were noted intraoperatively.\nHis immediate postoperative course was uneventful. He experienced intermittent knee swelling that was activity related, but this was not associated with inability to bear weight or other signs concerning for infection. However, 4.5 months postoperatively, he developed acute anterior tibial swelling with associated warmth and erythema at the tibial tunnel site. This was lanced at an outside facility after they suspected it to be an abscess, and he was placed on oral clindamycin and trimethoprim-sulfamethoxazole (TMP-SMX). He presented to our clinic 5 days later with persistent drainage from the wound and exposed tenodesis implant in the wound.\nHe then underwent formal debridement and irrigation with tenodesis implant removal and primary wound closure in the operating room. Gram stain from intraoperative specimens demonstrated few neutrophils, but no organisms. Infectious disease was consulted, and empiric intravenous vancomycin and piperacillin/tazobactam were started. However, by postoperative day 4, cultures grew N nova. Empiric antibiotics were stopped, and he was transitioned to oral TMP-SMX and intravenous meropenem.\nHe was doing well until 4 weeks after the debridement when he developed erythema, drainage, and wound dehiscence. He also developed an acute renal injury, so sulfamethoxazole-trimethoprim was switched to minocycline per infectious disease recommendations. He underwent a repeat debridement where a brownish fluid collection had developed around a braided suture tied to the end of the ACL graft. The suture was excised and wound thoroughly debrided. A diagnostic arthroscopy was then performed to rule out an intra-articular source of infection. It demonstrated an intact ACL graft and no evidence of the infection spreading from the tibial tunnel into the joint. Cultures taken at this debridement showed no growth, and he was transitioned to oral clarithromycin alone for 6 months. His wound healed without issues. His knee pain resolved, and he was able to return to his previous level of activity playing recreational basketball. |
A 25-year-old female presented to the emergency department (ED) for evaluation of persistent productive cough of yellowish sputum over the last four week and mild exertional dyspnea over the last two years. Her past medical history was unremarkable and she took no regular medications. There was no personal or family history of multiple endocrine neoplasia type 1 (MEN1) syndrome. She was in no distress on presentation to the ED with a resting hemoglobin oxygen saturation of 97% while breathing room air. Her physical examination was remarkable for absent breath sounds and decreased tactile fremitus on the left middle and lower lung fields. No wheezing or stridor were heard. Laboratory data were within normal limits.\nA chest x-ray (CXR) in the ED demonstrated opacification of the left middle and lower lung fields, hyperinflation of the right lung and deviation of the trachea to the left (Fig. ). A computerized tomography (CT) scan of the chest showed complete left lung atelectasis due to a mass obstructing the left main bronchus and excessive mediastinal deviation to the left with substantial herniation of the hyperdistended right lung into the left hemithorax (Fig. ). There was no evidence of tracheobronchial narrowing in the right lung or esophageal compression. The mass was well demarcated and of soft-tissue quality, demonstrating homogeneous contrast enhancement, starting 2.8 cm distal to the main carina, measuring 4.4 × 2 × 2.8 cm (Fig. ). Abdominal and head CT scans showed no abnormal findings. The patient subsequently underwent a diagnostic flexible bronchoscopy which revealed a pale hypervascular polypoid mass completely obliterating the left main bronchus which was biopsied using forceps (Fig. ). Histopathological examination of endobronchial biopsies disclosed a carcinoid tumor with a Ki-67 index of approximately 10%.\nFollowing thoracic surgery consultation, an open left pneumonectomy with concurrent complete lymph node assessment and dissection was performed. During surgery, the left lung was found completely atelectatic with adhesions between the pericardium and the left pleura which were dissected. No attempt of repositioning the mediastinum or placement of tissue expanders was performed, due to the absence of airway compression in the right bronchial tree during previous bronchoscopy and CT scan. The patient recovered well after surgery and no complications were noted. Post-operative histopathology disclosed an atypical carcinoid with a Ki-67 labelling index of 10% but no areas of necrosis (Fig. ). There was a radical resection of all tumor with clear operative margins, the periphery of the left main bronchus was infiltrated by tumor, but there was no invasion of the visceral pleura, and no infiltration of resected lymph nodes from lymph node stations 5, 7, 9 and 10 by carcinoid cells.\nPre-operative spirometry was as follows: FEV1: 1.51 lit (44% predicted), FVC: 1.54 lit (39% predicted), FEV1/FVC: 98%. Spirometry and static lung volumes 12 months after surgery were as follows: FEV1: 1.93 lit (58% predicted), FVC: 2.34 lit (61% predicted), FEV1/FVC: 82%, TLC: 3.28 lit (63% predicted), RV/TLC: 118% predicted. Although spirometry appears to be significantly improved after surgery, spirometry before surgery triggered fits of coughing and therefore preoperative values might not be representative.\nPostsurgical follow-up has included the following: Initial chest CT scan was carried out 2 months after surgery. Parathyroid hormone (PTH) and prolactin levels were within normal limits 1 year after surgery. The following investigations were carried out at 6 months and then every 6 months for the first 5 years: Chest CT scan, abdominal ultrasound, chromogranin A measurement and standard laboratory testing including complete blood count, renal function, liver function, calcium and glucose. Abdominal CT scan and fiberoptic bronchoscopy were carried out 1 year after surgery and then will be carried out annually for the first 5 years. Bronchoscopy would be performed earlier for any symptoms or imaging findings suggestive of local progression. Repeat chest CT scans after surgery showed no changes in mediastinal rotation compared to those prior to surgery, and no signs of tracheobronchial or esophageal compression. Repeat bronchoscopy showed a normal-appearing surgical stump of left main bronchus and no airway compression of the right bronchial tree. The remaining studies listed above have been normal. The chronic mild exertional dyspnea reported by the patient before surgery completely resolved on hospital discharge, 7 days after pneumonectomy. The patient has been asymptomatic for the last 16 months after surgery with excellent performance status. |
A 16-year-old female patient, complains of the appearance of her anterior teeth (abnormal anterior deep crossbite resembling monkey’s bite), with both upper and lower incisors being severely retruded and supraerupted. She was diagnosed with pseudo Class III malocclusion; the incisors are in an edge-to-edge relationship, but due to incisal interference between the upper and the lower incisors, an anterior functional mandibular shift was created and the teeth fall in deep anterior crossbite as she closes in centric occlusion. Skeletally, she was Class I; hence, this confirms the pseudo Class III malocclusion diagnosis ().\nIn order to construct the removable appliance “Hawley appliance with inverted labial bow,” the bite registration was taken for the patient in centric relation; this was carried out by guiding the mandible posteriorly in an edge-to-edge incisor relationship. The bite was then transferred to the working model articulated in a hinge articulator (). Construction of inverted labial bow was performed on the lower arch and 2 Adams clasps on upper first molars, and these represents the retentive components of this removable appliance, whereas, the active component was the helical Z bow spring fabricated behind the upper incisors and they are supported by a horizontal wire extensions and all these components were attached to the palatal acrylic plate, which was cut intentionally away from the upper anterior teeth ().\nTreatment of this case was carried out in 2 phases. In phase I treatment, the removable appliance with the inverted labial bow was used. The objective of this phase was to create a positive overbite and overjet and this was accomplished in one month. At the beginning, to test if the appliance was fabricated correctly, it was fitted passively in the mouth. This was performed by fitting the Adams clasps in the upper part of the appliance on the upper first molars and the acrylic plate on the palate with the helical Z bow spring passively lying behind the upper incisors, then her mandible was guided to bite in centric relation (edge to edge relation) placing the lower anterior teeth into the inverted labial bow in the lower arch. After making sure that the appliance was fitted correctly, it was then taken from patient’s mouth and 2 modifications were carried out before re-inserting it back. The first modification was expanding the labial bow by opening slightly the U shape of the inverted labial bow to fit it loosely on the lower incisors and the second modification was activating the helical Z bow spring by opening slightly the helixes of the spring to push the upper incisors forward out of crossbite. She was trained on wearing and removing the appliance several times, and instructions were given to her to use it all the time including nighttime. It will be removed only during eating and brushing. This was continued for 3 weeks. A follow-up appointment was made after 3 weeks of wearing the appliance, it was noticed that she was able to close in centric occlusion with positive overbite and overjet with no anterior forward shift. For retention, she was asked again to re-use the appliance at bedtime for another week and then the appliance was discontinued (). In phase II treatment, fixed appliances (braces) were placed to align teeth and to have proper overbite and overjet, which was accomplished in 11 months (). She had a very small size upper right and left second premolars; however, the upper right second premolar had undermined deep carious lesion that was extended to the roots, and it was hopeless to treat; thus, it was extracted before orthodontic treatment. The decision was made to mesialize the upper right first and second molar into its place. Asymmetrical extraction was also carried out in the lower right arch because even after having positive overbite, she still has Class III canine relationship on the right side and Class I canine relation on the left side. Thus, it was decided to extract the lower right first premolar during orthodontic treatment to correct the right side canine relationship and help to flatten the curve of Spee. Panoramic views of the case pre and post phase II treatment is shown in . Lateral cephalometric radiographs of the pre-treatment in centric occlusion, the pre-treatment in centric relation, and the post-treatment are shown in . The lateral cephalometric analysis of the pre-treatment measurements in centric occlusion (Pre-CO), the pre-treatment measurements in centric relation (Pre-CR), and the post-treatment (POST) measurements were compared with the mean cephalometric measurements in a normal adult of black ethnic race were shown in . Post-treatment measurements were almost normalized compared with the mean. Overall comparison of pre, progree, post-treatment extra-oral photographs were shown in , and intra-oral views were al gathered in . |
A healthy 8 year-old boy was referred to the endodontic clinic for the treatment of traumatically avulsed maxillary left central incisor after a car accident. The tooth had been found 4 hours after the accident. The upper right central incisor tooth also suffered from luxation.\nThereafter, the tooth was replanted by a general practitioner after 30 min. The dentist rinsed the socket and tooth with saline and prophylactic antibiotic therapy had been prescribed for one week. The patient was examined clinically and radiographically. An intraoral examination showed no laceration, abrasions, and contusion of the soft tissues. Maxillary central incisors were wire splinted with acid etch composite resin attached to the adjacent teeth for one week.\nThe next day, the replanted tooth was endodontically treated. The canal was instrumented and irrigated with normal saline and 2% sodium hypochlorite solution, alternatively. Calcium hydroxide paste (Merk, Darmstadt, Germany) was placed in the root canal (). In order to gain coronal seal, the tooth was filled with composite resin (Charisma, Kultzer, Germany). The calcium hydroxide paste was replaced three times during a year (3, 6, and 12 months). One year later the apexes of both central incisors were closed ().\nThe maxillary central incisors were radiographi- cally and clinically examined during scheduled appointments after the first year (). After one year the calcium hydroxide paste replaced for the last time and it was remained in the tooth. The tooth was restored with composite resin permanently. During the treatment, the vitality of right central incisor has been checked.\nAfter 5 years, the avulsed tooth kept in functional position. However, the avulsed tooth is infra- position because an ankylosis interferes with the vertical growth of the alveolar process. Although the space of pulp canal of the right central incisor is as wide as the first years, the growth has been completed; the apex is closed and the tooth remains vital (). |
A 38-year-old Caucasian woman, 35 weeks into her first pregnancy, presented to the emergency department for acute right-sided hip pain which precluded weight-bearing. Her right leg was shortened and externally rotated - there was no bruising or evidence of trauma.\nThe patient’s history was significant for hereditary thrombophilia (Factor V Leiden) and secondary anemia. Hip radiography revealed an unstable, displaced, right-sided femoral neck fracture with no evidence of osteonecrosis (Figure ). The decision to administer radiography, in this case, was based on the American College of Radiology guidelines, which cite an absence of in-utero deterministic effects of ionizing radiation effects after 27 weeks of gestation. Unfortunately, it was not possible to evaluate the symptoms of the patient with MRI at this time due to the coronavirus disease pandemic-induced stress on the healthcare system of our country.\nThe patient denied falls or trauma during the pregnancy, nor was there any history of smoking, alcohol abuse, use of glucocorticoids, or presence of rheumatologic/oncologic disease. Additionally, the patient was not malnourished, she underwent routine antenatal care, and took multivitamins. Serologic tests for inflammatory markers, as lab tests for serum calcium, phosphate, alkaline phosphatase, parathyroid hormone, vitamin D, and D-dimer returned normal.\nDuring multidisciplinary rounds, it was decided that delaying surgery was the best course of action out of fear of causing either mechanical or fluoroscopy-induced damage to the fetus during total hip arthroplasty. Five days later the patient experienced premature rupture of membranes, which was managed with emergency cesarean section (C-section) - no complications were encountered and a healthy 2300 g female was successfully delivered. Three days later the patient was transferred to our orthopedic surgery department for the treatment of the fracture. The significant degree of displacement (grade IV) of the fracture lasting over one week precluded open reduction with internal fixation due to fears of femoral head necrosis. During our literature review, we encountered a similar case of femoral neck fracture with grade IV displacement that was treated with open reduction internal fixation - despite restoration of blood flow to the femoral head within 15 hours, the authors still encountered femoral head necrosis with collapse six months later []. Given the considerable delay between symptom presentation and treatment, we decided the case warrants total hip arthroplasty instead of native hip salvage. Hemiarthroplasty was considered but was ultimately discarded as the conversion rate to total hip arthroplasty in young patients remain relatively high and the fracture was subsequently treated with a total uncemented prosthesis (Figure ), consisting of a 50 mm cup with 32 mm ultra-high-molecular-weight polyethylene insert and a 32 mm head with a 4 mm ceramic insert (Link Inc., Hamburg, Germany). Postoperative radiography confirmed prosthesis placement (Figure ); antibiotic and anticoagulant prophylaxis was initiated with ampicillin/sulbactam and enoxaparin sodium, respectively.\nThree days after the intervention the patient developed moderate abdominal pain without fever and accelerated intestinal transit, which both worsened over the next four days. Given the clinical presentation, there was a high index of suspicion for infection with Clostridium difficile (C. diff), although the diagnosis was ambiguous as the enzyme immunoassay (EIA) for the C. diff-specific antigen glutamate dehydrogenase was positive, while EIA for exotoxin A and B were negative. Due to exacerbation of symptoms and development of moderate hypokalemia (2.9 mEq/L), empiric treatment with metronidazole was began and marked rapid improvement. The patient was discharged five days later. |
A 60-year-old man with a past surgical history of C5–C7 anterior arthrodesis for cervical fracture 17 years ago was found on the floor after an unwitnessed ground-level fall, resulting in a head strike. He was taken to the local hospital in an Aspen collar with a GCS of 15 and no neurologic deficits. Routine ECG and laboratory evaluation was unremarkable except for elevated serum alcohol level. Full cervical spine computed tomography (CT) scan was performed which showed an Anderson and Montesano classification (20) type III left occipital condyle fracture (), a Levine and Edwards classification (21) type III (Jefferson) fracture with mild lateral subluxation of bilateral C1 masses (), and an asymmetric widening and slight anterior subluxation of the right AOJ (). A head CT was performed which showed no acute intracranial abnormalities.\nThe patient was immediately transferred to a level I trauma center for further management. In the emergency department, he reported severe midline neck pain, worse with movement, and unchanged paresthesia to bilateral upper extremities which he states is his baseline. Physical exam was unremarkable except for a right forehead hematoma with overlying abrasions and tenderness to palpation over the midline posterior neck. He exhibited full strength and unchanged baseline sensation to all extremities. He denied bowel or bladder incontinence and had strong rectal tone and intact perianal sensation. Through his clinical course, he became increasingly altered and agitated, refractory to medication. He was unable to maintain spinal precautions due to his agitation, and in an attempt to protect his cervical spine, he underwent endotracheal intubation.\nMRI of the cervical spine was performed and showed ligamentous injury at the craniocervical junction (CCJ) (Figures and ), asymmetric left odontoid-lateral mass widening, widening and subluxation of the right AOJ (), and edema of the paravertebral soft tissues around the fracture. CT and MRI established the diagnosis of OCD in a neurologically intact patient. Due to the unstable nature of the fracture, the patient was taken to the operating room for emergent occiput-C2 posterior instrumentation and fusion ().\nAn Aspen collar was in place at all times until surgical stabilization. Immediately, after removing the collar, Mayfield tongs were applied. Prior to patient positioning, baseline somatosensory evoked potentials (SSEPs) were obtained. Using a Jackson table turning frame, the patient was rotated into prone position. Fluoroscopic imaging was used during manual manipulation of the Mayfield apparatus to ensure no further displacement of the fracture.\nOcciput to C2 posterior instrumented arthrodesis was performed. Neuromonitoring remained stable during the procedure. The patient was discharged to home five days after presentation. |
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